The osteoclast-like giant cell is a benign tumor that presents as either an isolated benign tumor or one with a carcinomatous component. This study aims to report a rare case of osteoclast-like giant cell tumor (GCT) of the parotid gland.A 67-year-old female presented with a painless left pre-auricular swelling of 2-month duration which increased in size gradually over that period. On examination, there was a firm, mobile mass with well-defined borders in the left parotid gland. Fine needle aspiration cytology showed a giant cell-rich lesion that was highly cellular and contained a large number of osteoclast-like multinucleated giant cells, with clusters of spindle and epithelioid cells. Total parotidectomy was performed. After the operation, the patient was sent for radiotherapy.The histogenesis and exact nature of this tumor are unknown although numerous ideas have been put forward. The most common clinical manifestation is a painless slow-growing tumor in the parotid area. Primary osteoclast-like GCT of the salivary gland might show concomitant benign or malignant neoplasms. There is also a “pure form” of the tumor that has no accompanying neoplasm.GCT of the parotid gland is a rare tumor. The histogenesis and nature of parotid gland GCT are not completely understood. The treatment of choice is total excision followed by radiotherapy. •Giant cell tumors of the parotid gland are rare tumors.•The most common clinical manifestation is a painless slow-growing mass.•It can be in a pure form or a mix of benign and malignant components. It leads to extensive bone destruction ,2. The a2Patient Information: A 67-year-old female presented with a painless left pre-auricular swelling of 2-month duration which increased in size over that period. The patient was diabetic and hypertensive, and hence, was taking anti-diabetic and anti-hypertensive medications. Her past medical history aside from that was not significant.Clinical Findings: On examination, there was a painless, firm, and mobile mass with well-defined borders in the left parotid gland. No skin changes or ulceration over the mass were observed.Diagnostic Assessment: Hematological examinations revealed HbA1c of 7.7%, a blood sugar level of 227mg/dl, and an inflammatory marker (CRP) of 3.18 mg/l. On ultrasound examination, there was a well-defined hypoechoic solid nodule of about 33 × 27*11 mm in the left preauricular region. The nodule was in contact with the ramus of the mandible and showed malignant characteristics - along with a 7mm short axis lymph node seen in the left infra-auricular region without pathological sonographic characteristics. No obvious pathological lymph node was seen elsewhere in the neck.Fine needle aspiration cytology (FNAC) was performed which showed a giant cell-rich lesion that was highly cellular and contained a large number of osteoclast-like multinucleated giant cells with clusters of spindle and epithelioid cells (stroma) and a few lobulated benign-looking acini of parotid glands.The patient was sent for contrast magnetic resonance (MRI) imaging of the neck which revealed a single well-defined lesion in the superficial lobe in the left parotid gland measuring about 33 × 20 mm and showing a heterogeneous signal on T1 and T2, suggesting mostly pleomorphic adenoma. The right parotid gland was normal with bilateral benign-looking cervical lymph nodes of 6mm in the short axis - with normal submandibular and thyroid glands. The pharynx and larynx were normal.Therapeutic Intervention: Superficial-deep parotidectomy was performed on the patient. The result of histopathological examination revealed a giant cell tumor of 3.4 cm without any lymph node involvement . The histogenesis of this tumor is unknown although numerous hypotheses have been put forward . We coulThe most common clinical manifestation is a painless slow-growing tumor in the parotid area . The patIn previous incidents, a link between primary osteoclast-like GCT of the salivary gland and concomitant benign and malignant neoplasms was reported ,12.There is also a “pure form” of the tumor that has no accompanying malignancy . BecauseThe parotid gland, mandible, or soft tissues around the parotid gland can all cause GCT in the parotid area. The so-called GCT of the parotid gland appears to share some features with other GCTs, hinting that they have a separate etiology.The first feature is the tumor's link to neighboring anatomical components, as shown intraoperatively because extra-parotid tumors appear to be a continuation of their tissues of origin, whereas parotid tumors are distinct from the parotid . FurtherBased on the expression of epithelial and histiocytic markers from mononuclear cells, as well as their higher coincidence with malignant findings, some investigators claim that GCT of the parotid gland has an epithelial origin and looks more like carcinomas . DespiteThe recommended treatment, despite the vague nature of the tumor, is total excision of the lesion with healthy tissue margins to reduce the likelihood of recurrence, particularly those with associated malignancy. For patients with poor resection margins or those who cannot undergo surgery owing to underlying problems, radiotherapy can be considered to play a role in the treatment of these lesions .In conclusion, GCT of the parotid gland is a rare tumor. The histogenesis and nature of parotid gland GCT are not completely understood. It differs from GCT of bone in that it is not a continuation of neighboring tissues and there is no reactive bone production. The treatment of choice is total excision followed by radiotherapy.None is found.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.N/A.Abdulwahid M. Salh: Surgeon performing the operation, final approval of the manuscript.Zana H. Hassan: literature review, writing the manuscript, final approval of the manuscript.Razhan K. Ali, Suhaib H. Kakamad, Berwn A. Abdulla, Fahmi H. Kakamad: literature review, review and final approval of the manuscript.Ari M. Abdullah: The histopathologist who made the diagnosis, final approval of the manuscript.Fahmi Hussein Kakamad is Guarantor of this submission.N/A.None to be declared.


Hemimegalencephaly, or unilateral megalencephaly, is a sporadic congenital brain malformation characterized by enlargement of a cerebral hemisphere due to an abnormal proliferation of neurons or glial cells. Hemimegalencephaly is part of a spectrum of disorders, increasingly referred to as mTORopathies, which arise as a result of dysregulation or hyperactivation of the mammalian target of rapamycin (mTOR)-signaling cascade resulting in less restricted cell growth and survival. The resultant cortical disorganization and enhanced neuronal excitability often manifest clinically in the form of seizures. Ultrasound and magnetic resonance imaging (MRI) are often used to characterize hemimegalencephaly. Typical imaging findings seen include diffuse unilateral enlargement of a cerebral hemisphere with overlying cortical malformation and ipsilateral dilation of the lateral ventricle.in utero MRI revealed a mass-like lesion in the frontal lobe without associated perilesional cerebral edema. Keying in on abnormalities within the overlying cortex was crucial in suggesting focal hemimegalencephaly as a leading diagnosis and distinguishing it from alternative diagnoses such as a neoplasm. Follow-up fetal MRI demonstrated the evolution of the cerebral abnormality and confirmed the diagnosis. Early diagnosis facilitated appropriate counseling of the parents and guided postnatal imaging and management.This paper will review an unusual case of focal hemimegalencephaly diagnosed on prenatal imaging. Initial  Hemimegalencephaly is a rare congenital hamartomatous brain malformation with diffuse or focal enlargement of a cerebral hemisphere as a result of neuronal or glial proliferation ,2. It isThe involved hemisphere may have focal or diffuse neuronal migration defects, enlargement of the ipsilateral lateral ventricle, white matter gliosis, as well as an abnormal gyral pattern such as agyria, pachygyria, polygyria, and polymicrogyria ,6-8. In Hemimegalencephaly is a sporadic disorder. The prevalence is distributed equally by sex and is believed to be 1-3 per 1,000 epileptic children . It is aIn this report, we review the prenatal imaging findings of focal hemimegalencephaly, a variant of hemimegalencephaly in which there is focal enlargement within a cerebral hemisphere.A 37-year-old primigravida presented for routine prenatal care and underwent a fetal ultrasound at 25 weeks’ gestation. The prenatal ultrasound demonstrated an absent cavum pellucidum with midline shift. There was a suggestion of possible enlargement of the right frontal lobe. Fetal magnetic resonance imaging (MRI) was recommended for further characterization of the abnormalities.A fetal MRI performed at 26 weeks’ gestation Figure  revealedThe patient and her husband were counseled at a multidisciplinary conference with a maternal fetal medicine specialist, neuroradiologist, and pediatric neurologist. Given the fairly focal abnormality, concern was raised for other pathologies; however, hemimegalencephaly was the leading concern. A follow-up fetal MRI to reevaluate was offered and the patient agreed.Subsequent fetal MRI performed at 36 weeks’ gestation Figure  demonstrThe infant was born at term by Caesarian section. Postpartum physical examination demonstrated mild left hemiparesis. MRI on day three of life Figure  revealedAn electroencephalogram (EEG) revealed multifocal right hemispheric epileptiform discharges and right frontocentral seizures which were either subclinical or possibly subtly clinical with left upper extremity waving and left hemibody stiffening. He was immediately started on levetiracetam, followed by phenobarbital, and then topiramate before seizure control was achieved.At the age of two months, he developed infantile spasms which were treated with adrenocorticotropic hormone (ACTH) and ultimately vigabatrin. Over the next few months, the patient was successfully weaned off phenobarbital but otherwise remained on the complex antiseizure regimen.Ultimately, the infant underwent a right hemispherectomy at eight months of age. Anatomic pathology results were consistent with a cortical developmental malformation. His remaining antiepileptic medications were gradually weaned off over the following year. The infant has remained clinically seizure-free since the surgery.This report illustrates a case of hemimegalencephaly that presented as a focal variant during the second trimester of pregnancy. Findings on prenatal ultrasound that should raise suspicion for hemimegalencephaly include unilateral ventriculomegaly, asymmetric cerebral hemispheres with midline shift in the occipital region, subtle irregular sulcation or cortical thickening, and increased head circumference in the 90th percentile ,8,10.On MRI, the abnormality was characterized by low signal on T2-weighted imaging which was likely a result of the high cellularity in the malformed region . The unuThe diagnosis of focal hemimegalencephaly can be difficult to make based on prenatal ultrasound alone. If there is evidence of unilateral ventriculomegaly on ultrasound, in utero MRI should be considered . On MRI,Other diagnoses for a mass-like lesion must also be considered on the differential. Specifically, a true mass or neoplasm may exhibit a similar appearance on T2-weighted MRI. Subtle overlying cortical irregularities on prenatal ultrasound and MRI can suggest the diagnosis of focal hemimegalencephaly over a true mass or neoplasm. Additionally, while a neoplasm would be expected to demonstrate mass effect as well as peritumoral cerebral edema, focal hemimegalencephaly may not ,11. FinaIt is important to have an understanding of the early prenatal appearance of the focal form of hemimegalencephaly to facilitate appropriate care of the mother and infant. Early suspicion and diagnosis on fetal MRI allowed for appropriate counseling and discussions with the parents, set expectations regarding potential outcomes including the risk of intractable seizures, and allowed for a planned delivery with postpartum neonatal EEG monitoring. Thus, timely radiologic diagnosis of hemimegalencephaly expedited management and subsequently reduced overall seizure burden, ultimately minimizing developmental abnormalities and optimizing long-term outcomes -14.This case is unusual due to the initial low signal on T2-weighted sequences and the central, mass-like appearance of the right frontal germinal matrix with a lack of more diffuse unilateral features that are often described and expected in hemimegalencephaly. However, subsequent prenatal imaging demonstrated evolution into more typical radiologic findings associated with hemimegalencephaly which was confirmed on postnatal imaging. For prenatal imagers, it is vital to recognize the potential initial appearance of early focal dysplastic hemimegalencephaly as an awareness of this prospective presentation ultimately allows for and facilitates appropriate maternal and neonatal care.


Juvenile polyps, typically localized in the rectum and sigmoid colon, are a common cause of pediatric bloody stool. An isolated small intestinal juvenile polyp is uncommon and generally difficult to diagnose. The first case of an isolated juvenile polyp diagnosed by abdominal ultrasonography before acute abdomen had developed and resected by double-balloon endoscopy is presented along with a review of previous reports including this case. A two-year-old Japanese boy was referred to our institute for further evaluation of anemia persisting from one year of age. Laboratory findings showed mild iron deficiency anemia and elevated fecal human hemoglobin (Hb) and fecal calprotectin values. Upper and lower endoscopic findings showed no abnormalities. Because the abdominal ultrasonography performed one year later demonstrated a 15 mm jejunal polyp, combined with a similar finding on small intestinal capsule endoscopy, this was diagnosed as an isolated lesion. The lesion was resected by cautery with double-balloon endoscopy and diagnosed as a juvenile polyp pathologically. All clinical symptoms disappeared, and all laboratory data improved after treatment, without recurrence for more than one year after the procedure. Abdominal ultrasonographic screening and the fecal calprotectin value led to the diagnosis and non-surgical invasive treatment of an isolated small intestinal juvenile polyp. A two-year-old Japanese boy was referred to our institute for further evaluation and treatment of protracted iron deficiency anemia. The anemia improved with iron supplements, but relapsed following cessation of the medication. Similar episodes were repeated frequently, and fecal human Hb level was also elevated. The patient had no significant personal or family history. At the first visit to our institute, the patient had no growth retardation and no significant abnormalities in vital signs. During the period of iron administration, no physical abnormalities were observed. Moreover, he had no significant external deformities and normal psychomotor development.The patient’s initial laboratory tests showed Hb 11.2 g/dL, mean corpuscular volume 80.5 fL, mean corpuscular Hb concentration 26.4%, total protein 6.1 g/dL, albumin 4.1 g/dL, immunoglobulin G 461 mg/dL, and ferritin 19 ng/mL. Fecal examinations showed fecal human Hb 215 ng/mL and fecal calprotectin 134 μg/g. Both upper gastrointestinal endoscopy and colonoscopy performed at two years of age showed normal mucosal findings. At three years of age, the patient was re-evaluated by abdominal ultrasonography (AUS) concurrent with capsule endoscopy (CE). Abdominal ultrasonography showed a movable mass lesion in the mid-abdomen, and color Doppler flow showed a blood flow signal toward the mass, leading to the diagnosis of a pedunculated polyp. The polyp was localized in the upper jejunum, with a diameter of approximately 15 mm, and multiple anechoic cysts in the parenchyma, suggesting a juvenile polyp a,b. A saOne month after CE, oral DBE was performed, and an isolated pedunculated polyp, identified 75 cm anorectally from the pylorus, was resected by cautery with a high-frequency snare b–d. The Juvenile polyps tend to be localized to the rectum and sigmoid colon and are also associated with iron deficiency anemia due to protracted bloody stool and intussusception involving polyps as the pathologic advanced lesions ,2. The pTherefore, a review of this disorder, including the present case, was conducted, examining clinical features, onset age, sex differences, treatments, and examination procedures. Searching in the PubMed database, seven cases were reported between 1981 and 2022, including the present case ,7,8,9,10The facts seem to suggest the diagnostic difficulty of this disorder preceding intussusception. Abdominal ultrasonographic screening is thus extremely valuable for patients with chronic iron deficiency anemia presenting at around one year of age and relapsing after withdrawal of iron supplements, because no standard criteria for the acceptable age to perform small intestinal CE have been established.In addition, elevated fecal calprotectin values and their utility as a diagnostic aid have been reported in colonic juvenile polyps . HestvikIsolated small intestinal juvenile polyps may be treated non-surgically without intussusception developing with immediate diagnosis of the polyp using abdominal ultrasonographic screening and fecal calprotectin appropriately to make the differential diagnosis of protracted iron deficiency anemia.


Spinal epidural hematoma (SEH) is a rare disease. Several pathologies have been described as a cause, including trauma, arteriovenous malformations, coagulopathies, and iatrogenic causes. Spontaneous spinal epidural hematomas (SSEH) are blood in the spinal extradural space without a known cause. The incidence of SSEH has been estimated as 0.1 per 100,000 per year. Herein, we report a case of spontaneous spinal epidural hematoma in the cervical spine.We report a 57-year-old male patient who presented with sudden axial neck pain associated with upper and lower extremities weakness. Symptoms were precipitated by coughing. MRI of the cervical spine revealed an extradural lesion compressing the dorsal aspect of the spinal cord from C4 – C7. He underwent urgent decompressive laminectomy and evacuation of the hematoma. •We report a rare case of spontaneous cervical extradural hematoma.•Patient presented with sudden neck pain and quadriparesis.•MRI findings were not easy to interpret and to give a final diagnosis.•Other causes were excluded.•Urgent decompression was done. Patient improved postoperatively. PatieIn compliance with the SCARE guidelines , we repo2A 57-year-old man presented with acute onset neck pain with rapidly progressive quadriparesis of 9 h duration. Symptoms were precipitated by solid coughing. His past medical history includes hypertension and a drug history of Aspirin.Neurological examination disclosed quadriparesis. There was no urinary incontinence, and the cranial nerves and cerebellar functions were normal. His vital signs were normal. Blood workup, including platelet count and coagulation profile were normal.An emergent MRI of the cervical spine was performed. The MRI  showed pProvisional differential diagnosis was discussed with the patient. Risks and benefits were addressed, and the prognosis was clarified.The patient underwent an emergency operation done by a senior neurosurgeon. We did a C4 to C7 decompressive laminectomy and hematoma evacuation. Grossly, a well-organized clot was found in the corresponding levels, extending posterio-laterally. No other pathologies were detected intraoperatively, such as vascular malformation. No obvious bleeding source was identified, and hemostasis was achieved easily.The histopathology analysis of the specimen was suggestive of a hematoma, with no evidence of abnormal blood vessels suggesting the presence of any vascular malformation.The patient was diagnosed with a spontaneous epidural hematoma, after exclusion of the presence of coagulopathy, and vascular malformation on gross histopathology, and with no history of trauma.Postoperatively the motor power in all the limbs improved within 24 hours. The patient was discharged four days after surgery without any complications. He was transferred to a rehabilitation unit for physical therapies.3SSEH is a rare neurosurgical pathology with an estimated incidence of 0.1 per 100,000 individuals . It can The source of bleeding is considered venous in origin due to the absence of sphincters in the spinal epidural veins, thus making them prone to bleed after pressure changes. This is correct when talking about thoracic or lumbar hematomas. Due to low venous pressure in the cervical spine, this theory is thought to be invalid. A second theory owes the bleeding from free epidural anastomotic arteries in connection with radicular arteries in the cervical spine ,15.MRI is the modality of choice for diagnosis. In the first 24 hours, hematoma usually appears isointense to the cord on T1WI and hyperintense and heterogenous on T2WI. Then hematoma usually becomes hyperintense on T1WI and T2WI during the next 48 hours. Chronic hematomas appear hypointense on both T1WI and T2WI ,16. 17]17]. RadiUrgent surgical intervention is warranted within 12–48 hours of symptoms onset . Outcome4Spontaneous spinal epidural hematoma (SSEH) is a rare condition. They are considered a surgical emergency. MRI is the modality of choice for diagnosis. Surgical intervention is the ideal method for achieving complete recovery. Non-surgical management is reserved only for asymptomatic patients. As in our case, spinal cord compression due to hematoma after urgent surgical intervention can benefit from rehabilitation. This may lead to significant improvement in neurological functions.Not commissioned, externally peer-reviewed.This study was approved by the Ethical Committee of Prince Hamzah hospital.This work received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.Salem Al-Dwairy: Diagnosed and treated the case, wrote the largest share of the repot.Ala l-Mousa: writing the paper and critical revision of the work.Jehad Fataftah: final approval of the version be published.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):The author accepts full responsibility for this work, had access to the data and controlled the decision to publish.A written informed consent was obtained from the patient for publication of this case report and accompanying images.All authors have no conflicts of interest to declare.


Spinal ependymomas are the most common intramedullary spinal tumor, with a large proportion containing a small intratumoral cyst. Although the signal intensity varies, spinal ependymomas are generally well-demarcated, are not associated with a pre-syrinx, and do not extend above the foramen magnum. Our case demonstrates unique radiographic findings of a cervical ependymoma with a staged approach to diagnosis and resection. The patient is a 19-year-old female who presented with a three-year history of neck pain, progressive arm and leg weakness, falls, and functional decline. MRI revealed an expansile dorsal and centrally located T2 hypointense cervical lesion with a large intratumoral cyst extending from the foramen magnum to the C7 pedicle. Contrasted T1 scans showed an irregular enhancement pattern along the superior tumoral border down to the C3 pedicle. She underwent a C1 laminectomy for open biopsy and cysto-subarachnoid shunt. Postoperative MRI revealed a well-demarcated enhancing mass extending from the foramen magnum to C2. Pathology revealed Grade II ependymoma. She underwent an occipital to C3 laminectomy with gross total resection. Postoperatively she experienced weakness and orthostatic hypotension that improved remarkably upon discharge. Initial imaging was concerning for a higher-grade tumor, with holocervical cord involvement and cervical kyphosis. Given concern for grade and possible extensive C1-7 laminectomy and fusion for resection, a smaller surgery involving drainage of the cyst and biopsy was performed. Postoperative MRI revealed regression of the pre-syrinx, improved tumoral definition, and improvement of cervical kyphosis. This staged approach spared the patient unnecessary surgical intervention such as extensive laminectomy and fusion. We conclude that in cases of a large intratumoral cyst in an extensive intramedullary spinal cord lesion, open biopsy and drainage followed by resection in a staged fashion should be considered. Radiographic changes from the first procedure may affect the surgical approach for ultimate resection. Spinal ependymomas (WHO grade II) are the most common intramedullary spinal tumors, comprising approximately 60% of glial spinal cord tumors in adults and 30% in pediatric patients . Almost Classic grade II spinal ependymomas, also referred to as the cellular type, are generally well demarcated and accepted to exhibit classic imaging patterns, including iso- to hypo-intensity on T1-weighted MRI, hyperintensity on T2-weighted MRI, and strong homogenous gadolinium (Gd) enhancement on contrasted images . HoweverThe majority of spinal ependymomas are associated with a cystic component, which can be divided into the following three distinct types: intratumoral cysts (ITCs), rostral or caudal (polar or satellite) cysts, and reactive dilation of the central canal (syringomyelia) . Early rCystic involvement is important to recognize early as cystic presence may alter MRI interpretation, cyst volume has documented proportionality to symptom duration and severity, and cyst size and location may affect recommended treatment . ClassicThis article was previously presented as a poster at the 2022 AACNS Annual Congress on September 6, 2022.A 19-year-old female with a past medical history of congenital hearing loss presented with a three-year history of neck pain, progressive arm and leg weakness, falls, and functional decline. She had developed myelopathic gait and bilateral upper extremity weakness, which was very prominent in the hands. MRI revealed an expansile dorsal and centrally located T2 hypointense cervical lesion with a large ITC extending from the foramen magnum to the C7 pedicle. T2 central hypodensity extended beyond the cyst to the level of the T1-2 disc space, concerning for either infiltrative tumor, pre-syrinx, or syrinx with surrounding edema Figures , 1B. ConSeveral operative considerations were relevant to this case. First, although GTR is the traditional treatment gold standard for spinal ependymomas, this outcome is not possible in the case of malignant tumors, and in benign cases, the potential benefit must be weighed carefully against the preoperative clinical deficits, as almost half of patients experience worsening of their long-term neurological status postoperatively . Second,Employing a staged approach to cyst drainage and tumor resection with this patient yielded more accurate diagnostic information, allowing our team to make informed treatment decisions. This method revealed that a lesion which, on initial imaging, was concerning for a high-grade tumor with holocervical cord involvement and cervical kyphosis, spanned only from C1 to C3. The patient was spared unnecessary surgical intervention such as extensive laminectomy and fusion, and GTR was achieved. Postoperatively, chemotherapy and radiation are not recommended for this patient, as these treatments are usually reserved for incomplete resections and higher grade (WHO grade III-IV) tumors ,12.In cases of large ITC in an extensive intramedullary spinal lesion, precise accurate imaging may be disrupted by the cyst, disruption in cerebrospinal circulation, and associated features including hemosiderin cap. Diffuse cord signal change in these tumors may be mistaken for tumor but pre-syrinx and peritumoral edema should also be considered. Open biopsy and drainage followed by resection in a staged fashion should be considered in these patients, as large, malignant appearing intramedullary tumors with extensive ITCs may appear lower grade after cyst drainage. Radiographic changes from the first procedure may affect the surgical approach for ultimate resection.


We present a case of a greatly enlarged giant ectopic parathyroid adenoma that weighed 43 grams, which was located in the posterior mediastinum of a 74-year-old man. The patient presented with generalized weakness and decreased level of consciousness. He was found to have elevated level of serum calcium (19.9 mg/dl), and a subsequent assay of parathyroid hormone (PTH) was greatly elevated (2234 pg/ml). We report the course of management and outcome of the patient and present a review of the literature on giant ectopic parathyroid adenomas in the posterior mediastinum. Parathyroid adenomas (PA) usually arise in any of the four parathyroid glands located in their usual anatomic location in the neck, close to the thyroid gland. In some instances, they can arise in ectopic locations including intrathyroidal, intrathymic, mediastinal, submandibular, and within the carotid sheath . Their u99mTc-sestamibi scintigraphy and chest computerized tomography (CT) scan revealed a markedly enlarged posterior mediastinal parathyroid adenoma that was excised through open thoracotomy. The tumor was paratracheal on CT scan, a location that was reported only twice for such tumor. When excised, the tumor was found between vena cava, azygos vein and the spine, a location not reported previously. We discuss the surgical approach for such tumors, and the difficulties in excising them and causes of repeated surgical interventions. We reviewed previously reported cases of ectopic posterior mediastinal giant PAs.We present herein a patient who presented to the emergency department with generalized weakness and decreased level of consciousness. Laboratory workup revealed hypercalcemia. This prompted the measurement of PTH which was significantly elevated. 3/cubic mm. ESR was elevated at 30 mm/H . Sodium, potassium, and chloride levels were normal. Random blood sugar was 136 mg/dl . Urea and creatinine serum levels were both elevated at 72 mg/dl  and 1.41 , respectively. He had elevated calcium level at 19.9 mg/dl . Phosphorus level revealed mild elevation at 5.2 mg/dl . Liver function tests were within normal limits. Parathyroid hormone (PTH) level was markedly elevated at 2234 pg/ml . Due to the marked elevations of calcium and PTH level, he was admitted to the hospital for further investigation and management of hyperparathyroidism.A 74-year-old male presented with generalized weakness, decreased level of consciousness, poor oral intake, ataxia, dizziness, and disorientation in April 2020. Complete blood count revealed elevated white blood cells at 15.1 × 10Radiological investigations revealed unremarkable thyroid and pelvic ultrasound studies. Brain computed tomography (CT) scan displayed no intracranial hemorrhage, a few bilateral periventricular white hypodensities and ischemic small vessel disease. Chest CT scan exhibited a 6 × 4 × 3 cm well-defined lobulated heterogeneous mediastinal right paratracheal mass suggestive of neoplastic process, including enlarged lymph node or lymphoma. The possibility of a giant mediastinal ectopic parathyroid adenoma could not be ruled out based on elevated serum levels of calcium and PTH. Both lung fields appeared grossly unremarkable.Due to the marked elevations of calcium and PTH level, he was treated with zoledronic acid 4 mg intravenously over 30 minutes, Lasix 80 mg intravenously, along with intravenous fluids. On the following day, calcium level dropped to 15.1 mg/dl. On his discharge, three days later, his calcium level was at 9.8 mg/dl.99mTc-sestamibi scintigraphy scan was done at another facility after discharge, and it demonstrated focal abnormal persistent uptake inferior to the right thyroid lobe and extending into the mediastinum, correlating to the site of the previously diagnosed right paratracheal mass lesion  were noted. Very rare clusters of fat cells are noted. Many cystic spaces of variable sizes are noted, some contained eosinophilic material and others were empty; all were surrounded by sheets or clusters of clear cells  is not a rare disease, and it frequently exists as a mild condition lacking the florid renal and skeletal manifestations observed in the earlier literature. This is related to the availability of measurement of serum calcium levels, resulting in an increase in the discovery of cases of hyperparathyroidism (HPT) . Data frThe size and weight of a parathyroid gland are frequently the only intraoperative determinants of abnormality. The normal weight of the parathyroid gland was found in one study of 100 previously healthy subjects who died suddenly and had a postmortem examination to be 42.6 ± 20.3 mg, with a range of 22-103 mg. Gland weight varied with age, increasing to a maximum in the 41-60-year-old age group . In a stth percentile or 3.5 grams [Giant PA is defined as those with a weight more than 3.5 grams, based on a study of 300 patients with PA who underwent parathyroidectomy for primary hyperparathyroidism. The median weight of PAs in that study was 0.61 grams, with a range between 0.05 and 29.93 grams. The giant adenomas were defined as weight ≥ 95The parathyroid glands develop from the third and fourth pharyngeal pouches in humans between the fifth and twelfth weeks of gestation. The inferior parathyroid glands originate from the third pouch along with the thymus and are named parathyroids III (PIII). The association of thymus and PIII had been labeled as the parathymus complex. In about 5% of cases, PIII is located in the chest within the retrosternal thymus, its posterior capsule, or in contact with major mediastinal blood vessels. A lower position can result in PIII to be in contact with the pericardium or the pleura .The distribution of the location of PAs can be widespread, located as high as the submaxillary triangle down to the pericardium in one large series of 400 patients from Massachusetts General Hospital, with 84 parathyroid tumors were located in the mediastinum, 67 (16.75%) in the anterior, and 17 (4.25%) in the posterior mediastinum .50 patients with primary hyperparathyroidism were managed at a hospital in Germany, and 56 parathyroid tumors were excised. 33 of these were eutopic, while 23 were ectopic: 10 intrathyroidal, 7 intramediastinal, and 6 retrosternal. The authors defined the intramediastinal tumors as those requiring sternotomy or thoracotomy for extirpation. They reviewed seven series dating from 1941 up to 1974 with a total of 2089 parathyroid tumors. There were 144 tumors located in the mediastinum, ranging from 1.4% to 21% (average 6.9%) of all parathyroid tumors in these series .In a review of 145 cases of primary HPT from Mexico, an ectopic parathyroid location was detected in 13 cases (9%). The authors stated that their patients with primary HPT related to ectopic adenomas had significantly higher serum calcium levels, as in the current case, and larger tumors than patients with eutopic parathyroid glands, and more frequent HPT-related bone disease [The location of ectopic PAs, whether they are giant or nongiant adenomas, in the posterior mediastinum is quite uncommon. In one series, 17 ectopic mediastinal parathyroid adenomas (MPA), none of which were giant adenomas, were located in the anterior mediastinum, and none was observed in the posterior mediastinum . A recen99mTc-sestamibi scintigraphy had a reported sensitivity of 80-90%. This radiological study was helpful in the current case, and the location of the MPA was determined preoperatively. Ultrasonography has not been proven to be helpful in MPA in contrast to cervical PAs where it is of value. CT and magnetic resonance imaging (MRI) may provide valuable data with regard to its anatomic location. MPAs were rarely diagnosed before surgical exploration, and most patients had mediastinal exploration only after failed neck exploration [A literature review (1980-2012) of MPA showed that the prevalence of ectopic parathyroids from 28% to 42% in autopsy series and from 6.3% to 16% in surgical series of patients operated on for HPT and even up to 45% in patients who went reoperation. The actual prevalence of MPA has been reported to be 6%-30%. They were reported to be associated with more severe clinical manifestations with higher calcium levels, which could be related to delayed diagnosis and localization. Preoperative localization studies are quit important before mediastinal exploration is initiated. Technetium 99mTc-sestamibi scintigraphy combined with single photon emission CT (SPECT)/CT and 18F-fluorocholine PET/CT (FCH-PET/CT), revealing that FCH-PET/CT offers an equal or even superior detection of MPA [The evolving technology in imaging had resulted in improvement in localizing the source prior to surgery. One study showed that advances in radiology are helping in localization of MPA. A comparison is made between n of MPA .99mTc-sestamibi parathyroid scan (SPS) with SPECT/CT were utilized in 53 patients with primary HPT, and revealed that SPS interpretation method correlated perfectly with the surgical finding in 48/53 patients (90.6%); it was on the correct side of the surgical finding but not the exact location in 3/53 (5.7%) and nonlocalizing in 2/53 (3.7%) [3 (3.7%) . SPS was3 (3.7%) .Since the earliest scholarly description of pathology of primary HPT by Castleman and Mallory in 1935, adenomas were considered to form the commonest underlying lesion. The two basic criteria for the diagnosis of adenoma were that the lesion be solitary, involving one gland and occasionally two, and that the lesion be surrounded by a rim of parathyroid tissue. Involvement of a single gland was considered to be the most important criterion, inasmuch as a rim of normal parathyroid tissue was demonstrable in only about 50% of the cases [Sporadic HPT is caused by a PA in the majority of cases. Histologically, PAs are well circumscribed, with a thin capsule, and is composed of nests and cords of cells with rich capillary network. The dominant cells are the chief cells. In a minority of cases, oxyphilic cells can dominate. Cystic degeneration can occur in PA, as in the current case. PAs are usually devoid of fat cells, but they show rarely the presence of a few collections of adipocytes as in our case .In a recent review of parathyroid pathology, it was estimated that 80% to 85% of cases of PHP is caused by parathyroid adenoma, followed by primary parathyroid hyperplasia (15%), and parathyroid carcinoma (5%). The authors stressed previously set histopathological criteria for the diagnosis of PAs, namely, being well circumscribed, encapsulated by thin fibrous capsule, and composed of chief cells with clear cytoplasm and round nucleus, arranged within delicate capillary network. If not totally absent, stromal fat is sparse as in the current case. A rim of atrophic normal parathyroid tissue is seen outside the capsule in about half of the cases. The absence of such rim of normal parathyroid tissue does not exclude the diagnosis of PA especially in large lesions, as in the case reported here, because large PA, including giant PA, may have outgrown the preexisting parathyroid tissue . ParathyIn a study of 574 parathyroid glands resected at Memorial Sloan-Kettering Cancer Center (MSKCC) in New York, there were 195 adenomas. The authors stated that the current definition of PA is no longer purely histologic (complete circumscription and a rim of parathyroid tissue at the periphery with lack of fibroadipose tissue), but rather includes the effect of gland removal, with intraoperative PTH decrease and subsequent return to normocalcemia and long-term cure .We searched the literature for reports of giant PAs in the posterior mediastinum and found twelve cases, including one case with two giant adenomas –34 as weMany of the patients who had giant parathyroid adenomas underwent previous surgeries or explorations for excision but were unsuccessful. One study from Mayo Clinic reported 38 patients with parathyroid mediastinal tumors out of 2770 patients who underwent operations for HPT, including four giant parathyroid adenomas. These patients with MPA underwent more than one operation for removal of the tumor. It revealed one previous operation in 26 patients, two in 10 patients, and three in one patient. Two patients had also unsuccessful mediastinal explorations in addition to previous cervical operations . In one A study of 88 patients was operated on for persistent or recurrent disease underwent 98 reoperations. Missed hyperfunctioning parathyroid gland was found on reoperation in normal (eutopic) position in 49 (55.5%) patients and in ectopic position in 39 (44.3%) patients, including 20 (22.7%) cervical and 19 (21.6%) mediastinal. The authors concluded that multidisciplinary treatment team cooperation at a referral center consisting of accurate preoperative localization, expertise in parathyroid reexplorations, and correct use of intraoperative adjuncts contribute to a high success rate of parathyroid reoperations . Another99mTc-sestamibi imaging had 53% for all abnormal glands and 67% for ectopic glands, compared with ultrasonography (57% and 50%) and computed tomography (55% and 50%). They conclude that patients with persistent HPT requiring reoperative parathyroidectomy need additional imaging to localize the culprit parathyroid gland [A third study of over 700 parathyroidectomies revealed 46 reoperative cases for persistent HPT, with most patients had undergone bilateral neck surgery and yet the abnormal parathyroid glands found at reoperation were present in the neck (91%), with only 7 of these were ectopic either within the thymus gland or the mediastinum. The authors compared the positive predictive value (PPV) of various types of radiologic imaging in these cases and found that id gland .99mTc-sestamibi scintigraphy confirmed the presence of a posterior mediastinal paratracheal ectopic giant PA that weighed 42 grams, which was excised through open thoracotomy. Posterior MPA are quite uncommon with only 14 cases previously reported in the literature. We reviewed these cases and discussed reoperations in HPT which is encountered frequently in ectopic PA as well as the role of radiological studies in localization of these tumors.In conclusion, we present herein a case of a giant PA that presented with deteriorating consciousness due to hypercalcemia. Laboratory workup and


Thoracic esophageal rupture due to blunt trauma is very rare. Moreover, there have been no reports of thoracic esophageal rupture due to blunt abdominal trauma without chest trauma.We describe a rare case of esophageal rupture due to blunt abdominal trauma in a young female patient. Operation was delayed due to a misdiagnosis of chylothorax, and esophageal repair was performed six days after trauma. Postoperative esophageal leak developed and was treated with esophageal stent. She was discharged two months after surgery without sequelae.It is important to consider esophageal rupture as a differential diagnosis even in patients with only abdominal trauma, when in doubt. Thoracic esophageal rupture due to blunt trauma is an extremely rare clinical condition associated with significant morbidity and mortality. It is mostly caused by blunt trauma to the chest or to the chest and abdomen. There have been no reports of thoracic esophageal rupture due to blunt abdominal trauma without chest trauma. Here, we report a case of delayed diagnosis of thoracic esophageal rupture due to blunt abdominal trauma, treated with esophageal stent after primary repair.A 29-year-old female patient was referred to our hospital due to abdominal crush injury by a reverse vehicle. She complained of dyspnea and abdominal pain. On admission, she was conscious. Her vital signs were as follows: blood pressure of 102/60 mmHg, heart rate of 135 beats/min, respiration rate of 20 breaths/min, and temperature of 38 °C. Focused assessment with sonography in trauma (FAST) was positive in the abdomen and left thorax. Chest computed tomography (CT) revealed pneumomediastinum, left lower lobe atelectasis, left hydrothorax and fluid collection in posterior mediastinum, surrounding mid to distal esophagus, and descending thoracic aorta without abnormality in bony thorax (Fig. She was referred to the Department of Thoracic and Cardiovascular Surgery on the 5th day of hospitalization for worsening of left lung field haziness and her condition Fig. A. ThoracThe incidence of esophageal rupture due to blunt chest trauma has been reported to be 0.001% . MortaliThe treatment strategy for esophageal rupture has not been established yet. Several studies have shown that early diagnosis and management are associated with good clinical outcomes and that the golden time for treatment is usually within 24 h . WhetherIndications for esophageal stent are usually divided into two groups. The first indication is malignant or benign dysphagia . The secIn our case, we missed the possibility of thoracic esophageal rupture because there was liver laceration required emergent treatment and chylothorax can develop after abdominal trauma without chest trauma. The patient’s condition was getting worse and total left lung field haziness led to surgery on the sixth day after trauma. According to the operative findings, most of esophageal mucosa was intact, primary repair was performed. However postoperative esophageal leak occurred due to delay in diagnosis and surgery. Considering the patient’s poor condition, future quality of life and the relatively small size of esophageal leak, redo esophageal repair or other surgery, such as esophageal exclusion and diversion, was inappropriate. So, we decided to treat postoperative esophageal leak with esophageal stent. Esophageal stent was maintained for six weeks without complications. No leaks or other complications remained after stent removal.This case report shows that, although rare, thoracic esophageal rupture can occur with only blunt abdominal trauma. Therefore, it is important to consider esophageal rupture as a differential diagnosis even in patients with only abdominal trauma, when in doubt.


Pelvic chondrosarcomas are a major clinical challenge since the only therapeutic approach available is surgical resection. Reconstruction after partial resection of the pelvis including the acetabulum or the hip joint is a laborious and rigorous surgical procedure. Numerous complications are associated with different reparative methods. Moreover, due to the anatomical complexities of the area, adequate surgical margins are difficult to achieve in many cases, which are closely related to the advent of local recurrence of the tumor. Several techniques for hip function restoration and skeletal reconstruction have been reported. The purpose of this report is to describe a novel pelvic reconstruction technique for PI-II resection that required a custom-designed implant. We present the case of a 61-year-old female patient with chronic pain in the gluteal region. The pelvis’s magnetic resonance imaging (MRI) showed an osteolytic tumor in the right iliac wing that compromises the acetabular roof. The diagnosis was a grade 2 central chondrosarcoma. Surgery included the reconstruction of the acetabulum by inserting two Schanz pins coated with hydroxyapatite, one in the iliopubic corridor and the other in the ischium. A supporting “pyramid” was built, unitizing both Schanz with cement, onto which an acetabular cage was inserted. The procedure was completed with a conventional total hip prosthesis. The patient presented an acute prosthesis infection, which positively responded to prompt surgical lavage and antibiotic treatment. After 10 months of follow-up, the patient remains free of infection, with weight-bearing as tolerated, without pain, and with excellent hip motion. No tumor recurrence has occurred. Medialization of the construct has occurred as expected, with no evidence of implant loosening. The technique used in this patient is novel, could be considered cost-effective, and has allowed the reconstruction of a functional hip. For resections of the acetabular area and preservation of the ischium and pelvic zones, this technique may be an acceptable option. Bone tumors of the pelvis represent 10%-15% of primary malignant bone tumors . ChondroPelvic chondrosarcomas represent a complex clinical and surgical problem. This is because these tumors are radioresistant and chemoresistant, and the only therapeutic approach available is surgical resection. Therefore, the intervention should proceed in a way that margins are kept free of tumor tissue, which poses an additional difficulty .For tumors located in the acetabular region  , internaWith advances in implantation techniques, oncology surgeons have gradually adopted prosthetic reconstruction of the pelvis after tumor resection . ReconstThe choice of a particular technique for hip joint reconstruction depends on prognosis, functional capabilities of the patient, and particular surgical scenario and type of resection. A systematic review analyzed the clinical outcomes of internal hemipelvectomy, comparing up to seven types of reconstruction techniques, with unique challenges and complications for each method . MoreoveThis manuscript describes a pelvic reconstruction technique after a type I-II internal hemipelvectomy, for which hydroxyapatite Schanz pins, cement, and an acetabular cage were used.We present a case of a 61-year-old female patient with a history of hypothyroidism and obesity and several months of pain in the right hip and buttock.Magnetic resonance imaging (MRI) of the pelvis showed an osteolytic tumor in the right iliac wing that compromised the acetabular roof, corresponding to the Enneking and Dunham PI-II classification Figure . ComputeA surgical biopsy of the lesion reported low-grade myxoid chondrosarcoma. After reanalysis by the Hospital Oncology Committee, an internal hemipelvectomy and hip reconstruction were planned.Surgical techniqueAn extensive ilioinguinal approach was performed. The resection included the right hemipelvis, from the cephalic acetabular half to the sacroiliac joint, leaving only the caudal half of the acetabulum in continuity with the pubic rami. Two hydroxyapatite-coated Schanz pins were inserted, one in the iliopubic corridor and the other in the ischium. A cemented pyramid was molded by unitizing both Schanz, with cement, in which an acetabular reconstruction cage  was implanted, and a 46-mm cemented plastic insert was placed Figure .An uncemented stem with a 36-mm head was placed in the femur, achieving a stable hip. The cemented “pyramid,” as a consequence of weight-bearing, was assumed to be finally supported by the sacral ala due to progressive medialization.Staphylococcus aureus, Enterobacter cloacae, and Enterococcus faecalis. This was managed by prompt surgical debridement and implant retention, with the addition of intravenous (IV) antibiotics vancomycin + imipenem. Six weeks of additional oral treatment was continued with ciprofloxacin + amoxicillin-clavulanic acid. After the completion of treatment, the surgical wound removed has persisted without fistulae nor discharge and with nearly normal laboratory parameters.Postoperative deep infection occurred, with the presence of After a 10-month follow-up, she was in good general condition, with a healed and healthy wound, and walking without pain with assistance Figure . X-ray aPelvic chondrosarcomas pose significant challenges for management, since only surgical resection, attempting tumor-free margins, remains the treatment of choice. The anatomical complexity of the pelvis increases the difficulties of surgery, as well as the numerous complications associated with attempts at functional reconstruction, especially in cases of acetabular resections  . DiverseClinically, our patient presented dull and constant pain, as most patients with this diagnosis ,3. RadicChemotherapy and radiotherapy do not have a role as effective adjuvants against chondroid tumors ,3,5. ExtPelvic tumors may affect more than one of the regions described by Enneking ,9. If thA novel reconstruction technique for bone defects in patients with periacetabular tumors is the manufacturing of 3D-printed (customized) prostheses. A mean MSTS score of 80% functionality and no postsurgical complications have been reported in a series . It is aThe case we presented corresponds to type PI-II resection, where the ilium and the proximal part and roof of the acetabulum were removed. As such, there is no remnant bone for cranial support of a device. The technique designed for reconstruction considered a custom-made prosthesis with hydroxyapatite Schanz, cement, and cage. The aim, such as with other methods, was to restore a functional and stable hip joint. The construct was designed to progressively medialize with weight-bearing, till supporting against the sacral ala. Of note is the fact that medialization led to a more vertical position of the cup, originally inserted at 40° of inclination. Being this a matter of concern for the fate of the construct, should this method be indicated in future cases, care must be taken to provide a nearly horizontal orientation for the cup. This may lead to a more favorable inclination angle when medialization is completed.The main limitation of the case presented here is the short-term follow-up. Complications such as tumor recurrence and/or mechanical failure, and infection are still possible.However, in the case of type II and I + II resections, it can be a valuable, low-cost, and cost-effective option, as it does not require sacrificing the limb and allows for satisfactory functional recovery of the hip joint in the short term. Utilization of the remaining bone corridors for hydroxyapatite-coated Schanz fixation, unitizing them with cement, in which a cage can be inserted, along with progressive support of the construct against the sacral ala, could be validated as an alternative to more costly methods. Allografts, allografts + prosthesis composites, and customized implants have been more extensively utilized. Nonetheless, complications that may lead to failure are still frequent with any of these. We see no reason indicating that the outcome associated with our method may be less favorable.In summary, we present a patient with low-grade myxoid chondrosarcoma that compromised the right iliac wing and acetabular roof (type I + II resection) who underwent a reconstructive surgical technique that preserved the limb and allowed a satisfactory functional recovery of the hip joint in the short term. The method used in this patient may be cost-effective, if, when evaluated in more patients and with longer follow-up, functional results and complications prove at least comparable to others.


A previously well 15-year-old male presented with a history of gross rectal prolapse (GRP) involving full-thickness rectal prolapse of increasing severity and incidence over 6 months that occurred with every bowel motion, varying from 10 to 40 cm. He denied constipation and passed a soft motion once daily, adeptly reducing his prolapsed rectum after each motion. This case illustrates technical challenges and planning for surgical intervention for optimal treatment in keeping with an FDA alert issued April, 2019 banning surgical mesh for pelvic organ prolapse. Preoperative fluoroscopic defecography confirmed rectal prolapse beginning with eversion of the anal verge identified on inspection. For surgery, general anesthesia was induced, he was placed in a Trendelenburg position, and four ports were inserted. The peritoneum was incised and blunt dissection used to expose the levator ani complex (LAC) taking care to prevent lateral nerve injury and preserve regional vascularity. Seven polypropylene sutures were used to fix the seromuscular posterior wall of the rectum to the median raphe of the LAC, the presacral fascia, and the periosteum of the sacral promontory. Operative time was 170 min. Postoperative recovery and progress were unremarkable. Currently, 5 years postoperatively, defecation is regular without recurrence of prolapse. For prolapse involving protrusion of the upper rectum without eversion of the anal verge, rectal fixation to the sacral promontory without further dissection beyond the peritoneal reflection is adequate, but when extensive prolapse is associated with eversion of the anal verge, more extensive blunt dissection from the peritoneal reflection to the LAC with multiple rectopexy sutures is valid for reducing risks for recurrence and eliminating mesh-related complications. Various procedures have been described for the repair of gross rectal prolapse (GRP) with varying success. Successful surgical intervention and technical aspects of repairing GRP in an adolescent are presented.A 15-year-old Japanese male presented with a 6-month history of recurrent full-thickness GRP. He was an only child, otherwise well  with no remarkable previous medical history; physical and mental status were normal; no specific family history or psychosocial issues were identified. He had no history of previous surgical interventions. Bowel function was regular  without constipation. Prolapse occurred with every bowel motion without any obvious cause and varied from 10 to 40 cm . He adepOn assessment, a trial of conservative management was recommended. Probiotics and laxatives were used daily to soften his stools to eliminate straining without any real improvement. After 6 months, fluoroscopic defecography was performed by filling the rectum with undiluted barium contrast agent and asking the patient to strain and expel the barium. Eversion of the anal verge was observed before more proximal rectum and sigmoid colon prolapsed on fluoroscopy . The extGeneral anesthesia was induced with the patient supine and four ports  were inserted, conventionally . The patDiagnosis, differential diagnosis, reasoning/indications for treatment and treatment goals, and prognosis were discussed with his caregivers according to CARE guidelines. Written informed consent was obtained from his caregivers for publication of his case report with images. A timeline figure was not included because diagnosis, treatment, and follow-up were straightforward.The trend for repairing rectal prolapse in the literature is minimal retrorectal dissection without breaching the peritoneal reflection, preservation of the posterior rectal space and lateral rectal ligament, and localized rectopexy to the sacral promontory, a maneuver reported to effectively control recurrence to a rate of around 5% , 2. An aFor preoperative planning, a defecography is mandatory despite concerns about radiation exposure . MagnetiDeep retrorectal dissection was successful as a mesh-free option for treating GRP.The original contributions presented in this study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.GM treated the patient and prepared the manuscript. SY and HM treated the patient. GL revised the manuscript. AY supervised the entire drafting of the manuscript. All authors have read and approved the final manuscript and contributed to the article and approved the submitted version.


Colloid cyst (CC) is a rare and benign cyst found in the third ventricle near the foramen of Monro. Although the role of surgical resection is well established in symptomatic large-sized CC, it remains debatable whether surgical removal of CC with no symptoms or minimal symptoms is necessary.A 49-year-old male patient visited our institute for incidentally detected intracranial mass. MRI demonstrated typical, 12 mm-sized CC located in the third ventricle. It was noticed that the cyst spontaneously decreased in size from 12 mm to 4 mm on MRI at 18 months after the first visit.Although spontaneous regression is a very rare phenomenon in CC, regular imaging study and frequent neurologic examination can be an alternative option for well-selected, asymptomatic cases. Colloid cyst (CC) is a benign cyst found in the third ventricle near the foramen of Monro . It is rAlthough CC is thought to be congenital, the pathogenesis of CC remains controversial . This cyA 49-year-old male patient visited our institute for an incidentally detected intracranial mass. The patient had no underlying disease. He did not complain of any symptoms such as headaches or dizziness. On neurological and physical examination, there were no specific findings or neurological deficits associated with intracranial hypertension. Brain MRI revealed a 12 mm-sized CC located in the third ventricle. The cystic lesion was a round and well-demarcated lesion located in the right foramen of Monro and the upper third ventricle. The lesion was isointense on T1-weighted images and predominantly hyperintense on T2 with a hypointense focus within the lesion. The signal intensity pattern was the same on fluid-attenuated inversion recovery (FLAIR) images likely due to the highly proteinaceous nature of cyst fluid. There was neither hydrocephalus nor any signs of hemorrhage. The patient was followed up with regular imaging protocol. It was noticed that the cyst spontaneously decreased in size from 12 mm to 4 mm on MRI at 18 months after the first visit. As a result of follow-up at 30 months, the size of the lesion was maintained constant without any clinical symptoms (Fig. CC is a pathologically benign intracranial mass . The cliThe present case is the tenth report on spontaneous regression of CC , 7, 9–15Although the pathophysiological cause of spontaneous regression has not been elucidated yet, a scenario in which the size decreases due to unrecognized cyst rupture is conceivable. Motoyama et al. were the first to report spontaneous regression of CC. They radiologically confirmed it as a cyst rupture. The cyst might rupture at an unknown time and all its contents could be absorbed into the ventricle, leaving only traces of the cyst wall in CC with no or minimal symptoms . All spoFor asymptomatic CCs, careful definition of the asymptomatic criteria is necessary because observation with follow-up imaging can be considered as a treatment option. Some patients say that they have no symptoms if the headache is minor, whereas others may report or feel it severe even if the headache is mild. In addition, if the size of CC increases, other signs of increased intracranial pressure should not be overlooked, and the patient should be educated.Surgery should be considered, especially if symptoms develop during observation with an increase in the size of CC. One analysis has suggested predictors of asymptomatic to symptomatic progression, such as younger age, increased cyst size, ventricular dilatation, and increased signal on T2-weighted MRI . HusseinIn conclusion, surgical resection is a main treatment for symptomatic or asymptomatic large CC. However, it sometimes regresses spontaneously without specific treatment. Therefore, well-selected asymptomatic CC can be observed with regular imaging study and frequent neurologic examination.


Surgical site infections are a common complication in the management of patients with solid cancer tumors in cytotoxic chemotherapy. The antibiotic combination chosen depends on the neoadjuvant therapy used and the anatomical site and extent of the surgery. In this brief report, we describe a surgical site infection caused by an unusual microorganism and a succinct review of the pathogen's behavior. Shewanella species are a group of bacteria commonly found in aquatic habitats. The first description of the species was in 1931 from water supplies of dairies. First known as Achromobacter putrefaciens, then Pseudomonas, in 1985, they were finally reclassified as a new genus, Shewanella [ewanella .Shewanella putrefaciens. Here, we present the first Shewanella algae surgical site infection, without previous common exposures or nosocomial outbreak association. Surgical site infections are an unexpected and unique presentation of Shewanella sp, with only few previous case reports, associated with A 74-year-old woman with sigmoid cancer, adenocarcinoma (T3N0M0), was admitted to our hospital with acute abdominal pain. The patient had a recent diagnosis of colorectal cancer and went to a scheduled surgery during the first week of December. She has a previous history of diabetes, hypertension, and fibrocystic breast disease. She is from an urban location in Panama City with no history of travel due to the COVID-19 pandemic and a recent diagnosis of cancer. She has no previous exposure to seawater or any marine environment exposure, neither a history of eating seafood nor fish.After careful preoperative assessment and control of her comorbidities, the patient underwent a scheduled lower anterior resection with colorectal anastomosis without immediate complications. On the seventh postoperative (PO) day, she returned to the emergency room with a 48-hour history of nausea, vomiting, abdominal distention, and pain. An emergency exploratory laparotomy was performed, where they found a 50% dehiscence of the colorectal anastomosis, together with fecal material that drained through the intestine into the pelvic cavity, with inflammatory material. Surgical dismantling of the colorectal anastomosis with terminal colostomy and cavity wash were done.Shewanella algae and Enterococcus faecalis, using matrix-assisted laser desorption/ionization and time-of-flight mass spectrometry (MALDI-TOF MS).She was admitted to the intensive care unit (ICU) where she had a torpid evolution with septic shock. Cefepime and metronidazole were initiated. During the fifth day after admission, she developed colostomy necrosis and was taken again to the operating room (OR) for colostomy revision where a parastomal abscess was found. After this, the patient remained stable until the 15th day PO, when the systemic inflammatory response syndrome (SIRS) signs appeared again. Space surgical site infection was suspected, and an abdominal computed tomography (CT) scan was done, which revealed intra-abdominal collections in the left paracolic gutter, right paracolic gutter, and pelvic cavity next to the rectal stump. The left paracolic gutter collection was drained percutaneously, and 700 ml of pus was collected Figures . The pusAntibiotic regimen was then switched to intravenous imipenem 500 mg twice a day (q12h) and vancomycin 1 g q12h (ampicillin was not available at the time). Due to the extremely unusual isolation of this bacteria and the implications in nosocomial outbreaks, the patient went under contact isolation precautions; the sterile solutions used for abdominal cavity wash were analyzed and colonization screening was implemented to the operating room staff, nonetheless the results were negative. Several hospitalized patients were tested for colonization, with negative results. Shewanella algae. The patient had a favorable clinical course and, after one month of hospitalization, was discharged. On the follow-up appointment, two weeks after the hospital discharge, she was afebrile with a functioning colostomy and had scheduled chemotherapy treatment plan. This is the first Shewanella algae associated surgical site infection reported in Panama, and the second Shewanella species associated surgical site infection case published in the English literature.Besides the antibiotic and surgical therapy, the wound was managed with vacuum-assisted closure (VAC) and finally resolved at the 25th PO. A culture was taken from the fluid of the VAC sponge on the 23rd PO which was positive for Shewanella species are Gram-negative, nonfermentative, motile bacilli. They are widely distributed in the environment and have uncommonly been associated with a variety of human infections [Shewanella disease in humans remains low compared with other pathogens, there are an increasing number of reports, probably because of better diagnostic tools.fections . They wefections . AlthougShewanella putrefaciens and Shewanella algae are the species often associated with human infections; however, S. haliotis and S. xiamenensis have also been reported [S. putrefaciens and S. algae because the latter species is not included in many databases [ S. algae from an abdominal fluid aspirate and later from a surgical sponge, thanks to matrix-assisted laser desorption/ionization time-of-flight mass spectrometry (MALDI-TOF MS).reported . Automatatabases ,4. In ouShewanella sp human infections was made in 2013, which reported 239 cases, none of them were surgical site infections [Shewanella putrefaciens after abdominal hernioplasty [ Shewanella species isolations from intra-abdominal sources, most of them are thought to be colonization [This strange organism has been associated with otitis externa, soft tissue infections, bacteremia, hepatobiliary infections, endocarditis, pneumonia, cerebral abscess, and meningoencephalitis -6. The mfections . To our ioplasty . There hShewanella infections are contact with seawater with a previously injured skin via recreational or occupational exposure and consumption of seafood (70% of cases) [Shewanella infections [Shewanella algae is susceptible to aminoglycosides, carbapenems, erythromycin, and quinolones [Shewanella species are susceptible to narrower spectrum antibiotics such as amoxicillin and third-generation cephalosporins; however, they can show resistance to imipenem by secreting oxacillinases [The major risk factors for f cases) . In a ref cases) . Conditifections . In our inolones . Many ShShewanella species, although an infrequent cause of infections, appear to have an increasing number of outbreaks and reports. Regarding surgical site infections, Shewanella is an extremely rare association, probably because the common entry mechanism is through injured skin in contact with water, fish or seafood consumption, or medical equipment contamination. It is important to count on new generation techniques such as MALDI-TOF MS to make the accurate identification with susceptibility to guide antibiotic therapy.


Lipoma is a common benign soft tissue lesion that can virtually develop anywhere in the body. However, the retropharyngeal space is an extremely rare location for lipoma. We report the case of a 48-year-old man who presented with progressive dysphagia of 3 months duration. It was associated with a weight loss of 6 kg. There was no history of cough, regurgitation, or heartburn. He was a heavy smoker, but he denied consuming alcohol. On examination, the patient was noted to have a hoarse voice. Examination of the oral cavity revealed a bulge in the posterior pharyngeal wall with intact and smooth overlying mucosa. Examination of the neurological system revealed normal findings. The patient was referred to undergo a computed tomography of the neck, which demonstrated a well-defined homogeneous lesion with fat density in the retropharyngeal space. Subsequently, magnetic resonance imaging revealed a well-encapsulated midline retropharyngeal mass, measuring 4.6 x 10.2 x 13.8 cm, filling the retropharyngeal space and extending from the C2 vertebra superiorly to the inferior border of C7 inferiorly. The mass has a high signal intensity on T1- and T2-weighted images with complete suppression of the signal on the fat-saturated sequences, likely representing a retropharyngeal lipoma. The patient underwent surgical resection of the tumor by the lateral cervical approach. Histopathological examination showed lobules of mature adipose tissue, representing a lipoma. Retropharyngeal space is a very rare location of lipoma. The case highlights the importance of considering retropharyngeal lesions when encountering a patient with progressive dysphagia. Lipoma is the most common benign mesenchymal tumor and can virtually develop anywhere in the body . It is eA 48-year-old man attended the primary healthcare center with a complaint of difficulty in swallowing for 3 months duration. Initially, he experienced difficulty swallowing certain solid foods. However, his difficulty in swallowing progressed to involve both solids and liquids. The difficulty in swallowing was associated with an unintentional weight loss of 6 kg in the last month. There was no history of cough, regurgitation, or heartburn. Furthermore, no history of painful swallowing or shortness of breath was given. Regarding his past medical history, he had a 20-year history of well-controlled hypertension, diabetes mellitus, and dyslipidemia. No history of peptic ulcer disease was given. The family history was remarkable for systemic lupus erythematosus in his two siblings. He was a heavy smoker (25 pack-years). He never consumed alcohol or used recreational drugs.2. He was noted to have a hoarse voice. Examination of the oral cavity revealed a bulge in the posterior pharyngeal wall with intact and smooth overlying mucosa. On neurologic examination, the patient had normal gag reflex and normal cranial nerves examination. There was no impairment of the motor or sensory function. No lymphadenopathy or palpable neck masses were observed. Laboratory investigations, including hematological and biochemical profiles, were normal. During the laryngoscopy examination, a marked protrusion of the posterior pharyngeal wall with narrowing of the hypopharynx was noted. The larynx appeared normal.On examination, the patient was alert, conscious, and oriented. The vital signs were within the normal range. He had a body mass index of 27.5 kg/mThe patient was referred to undergo a computed tomography (CT) scan of the neck, which demonstrated a well-defined homogeneous lesion with fat density in the retropharyngeal space Figure . SubsequThe clinical diagnosis and the management were discussed with the patient and afterward, the patient agreed to undergo surgical resection of the tumor. The surgery was conducted under general anesthesia using the right lateral cervical approach. Careful dissection was performed to expose the lesion, which appeared as a large yellowish mass. The lesion was excised completely. The recovery was uneventful. Histopathological examination showed lobules of mature adipose tissue, representing a lipoma Figure .Postoperatively, the patient had significant improvement in his swallowing. The patient was discharged on the fourth postoperative day. At the 4-week follow-up visit, the patient regained normal swallowing and experienced weight gain.We report a rare case of a giant retropharyngeal lipoma that underwent successful surgical excision. The retropharyngeal space is a potential space in the neck that spans from the skull base to the mediastinum. It is bounded by the buccopharyngeal space anteriorly, and the prevertebral space posteriorly . PrimaryWe conducted a review of the literature published from 2000 to 2022, by searching PubMed, MEDLINE, and GoogleScholar databases. A total of 27 cases were identified, which are summarized in Table Since the retropharyngeal space is not accessible for clinical inspection, cross-sectional imaging is vital to establish the diagnosis of retropharyngeal lipoma. On CT, the retropharyngeal lipoma appears as a well-circumscribed non-enhancing lesion with homogeneous fat attenuation . FurtherRetropharyngeal space is a very rare location of lipoma. The case highlights the importance of considering retropharyngeal lesions when encountering a patient with progressive dysphagia. Since the retropharyngeal space is inaccessible to clinical inspection, cross-sectional imaging modalities are essential to make the diagnosis. Surgical resection is curative and the transcervical approach is safe and feasible for the resection of a giant retropharyngeal lipoma.


The incidence of snapping popliteus tendon syndrome, a type of lateral knee snapping, is not high, so making an accurate diagnosis is difficult. A proper treatment following an accurate diagnosis is essential for improvement. Very few cases have been reported of its treatment.An 18-year-old male patient had experienced painful popping in the lateral part of the knee during knee flexion for 3 years before his hospital visit.Snapping popliteus tendon syndrome.The patient underwent an all-arthroscopic surgery. Tendon debulking and tissue debridement around the popliteus tendon was conducted, but the snapping did not resolve. The enlarged tubercle was excised through an arthroscopic procedure using a burr, and the surgery was finished after confirming that snapping was resolved.Full range of motion (ROM) was recovered 6 weeks after surgery and the snapping did not recur.Snapping popliteus tendon syndrome is a disease that is hardly recognized due to its low prevalence and difficulty in diagnosis, and it requires close observation of the patient before surgery. The location of the tenderness and the snapping occurrence must also be carefully identified. Our procedure is an entirely arthroscopic technique; as it has the prominent advantage of a speedy recovery and easy rehabilitation, it could also be helpful to set treatment standards for this disease in the future. A proper treatment following an accurate diagnosis is essential for improvement. Accordingly, caution is required for the diagnosis and treatment of this condition. There have been 6 case reports of snapping popliteus tendon syndrome so far,–8 and the last report was the case reported by Krause et al in 2008. Recently, a case of cyamella in the popliteal tendon, which causes snapping and discomfort, has been reported, but that was a special case and could be considered a disease with a different pathophysiology. Arthroscopy has been frequently used for the diagnosis of snapping popliteus tendon syndrome, but the treatment was often conducted as an open procedure. In this case report, the diagnosis and treatment of snapping popliteus tendon syndrome were all conducted by arthroscopic procedures, and it was successfully treated. Accordingly, the results of the case study of snapping popliteus tendon syndrome are reported here.Lateral knee snapping is a rare disease; patients who experience lateral knee snapping may hear a popping sound or feel a snapping sensation during a specific motion and the condition is often accompanied by pain. There are various causes of lateral knee snapping, including iliotibial band friction, biceps femoris tendon snapping, popliteomeniscal fascicle tear, popliteus tendon snapping, etc; as the symptoms of this condition are not specific, it is often difficult to differentiate it from other diseases.http://links.lww.com/MD/H743, this video was taken before surgery, and a clear popping sound can be heard). No specific findings were shown on plain radiography, and there were no abnormal findings of the meniscus or cartilage on magnetic resonance imaging (MRI). Inflammatory changes around the popliteus tendon may have been responsible for the popping, but the etiology was not clear . After surgery, range of motion (ROM) and quadriceps strengthening exercise were performed while the patient received adequate pain control; full ROM was recovered 6 weeks after surgery and the snapping did not recur .An 18-year-old male patient had experienced painful popping in the lateral part of the knee during knee flexion for 3 years before his hospital visit. He reported no history of trauma. The patient said that he had been a football player until high school and that when the painful popping got worse, he stopped playing football. However, his symptoms had not improved. No other specific findings were shown on physical examination, and we heard a clear popping sound during knee flexion and extension (see Video 1, Supplemental Video 1, ear Fig. . The enl,11 The popliteus muscle is known to play an essential role in the knee posterolateral anatomic relationship. According to LaPrade et al, it is located anterior to the popliteal sulcus from full extension to 112º flexion and is located in an anteriorly subluxated position. Additionally, it is engaged into the popliteal sulcus from 112º flexion to full flexion.The popliteus muscle originates from the posterior surface of the proximal tibia, passes through the popliteal hiatus as a tendon, and attaches to the lateral femoral condyle. It is a tendon with a fairly wide footprint on the lower anterior surface of the lateral epicondyle and the proximal half of the popliteal sulcus with a complex structure that is connected to the lateral meniscus by popliteomeniscal fascicles as it passes through the popliteal hiatus.Although it is slightly different across individuals, engagement occurs into the popliteal sulcus after a certain angle of flexion. Before engagement, snapping sometimes occurs in the tubercle located below the popliteal sulcus in the mid-flexion state. Case reports have shown that snapping occurs mostly at 20º to 40º of flexion (Fig.  Mariania et al also reported positive findings for Cabot’s sign, which is almost the same test. However, this type of physical examination has a somewhat low specificity because it may not be clearly differentiated from pathologic signs of other lateral compartment structures, such as fibular collateral ligament, or medial compartment structures. Furthermore, it is difficult to discuss the sensitivity of the test due to the insufficient number of cases. Therefore, its use is considered limited.Snapping popliteus tendon syndrome is a rare disease; only 6 cases have been reported so far. It is difficult to diagnose as no specific findings were shown on plain radiography or MRI. Cooper et al conducted a physical examination to confirm popliteus snapping when performing flexion-extension ROM while applying varus stress in the “figure of four” position, and all 6 patients showed positive findings.As shown above, snapping popliteus tendon syndrome is a condition that is difficult to diagnose based on imaging tests or physical examination alone; the use of arthroscopic examination is helpful to diagnose and treat this condition. In this case, we performed an arthroscopic examination and could confirm that snapping occurs, and the snapping was treated with all arthroscopic procedure. Debulking of the hypertrophied fibrotic tendon and surrounding tissues was carried out, and the enlarged tubercle was excised using a burr because the snapping had not resolved after debulking.Various treatments have been introduced in the case reports so far, and there were many cases of operation with the open procedure after diagnostic arthroscopy. In these 6 case reports, surgeries could be classified into 3 procedures: popliteal tendon release or debulking, tenodesis with or without sulcus deepening, and tubercle excision Table .,14 Some cadaver studies have shown that isolated popliteal tendon resection did not induce posterolateral instability. However, clinically, resection may affect some of the various functions of popliteus. Therefore, it should be considered as the last method when snapping is not resolved even after treatment with other methods.The way to resect the entire popliteal tendon can completely resolve the occurrence of snapping, but it has the disadvantage of removal of the function of the popliteus. This may induce posterolateral and rotatory instability and may affect meniscus withdrawal during flexion.Techniques such as tenodesis with fibular collateral ligament or tenodesis after sulcus deepening procedure cannot be performed with arthroscopic surgery alone. Since invasive open surgery is required, this procedure has the disadvantages of a poor recovery after surgery, prolonged rehabilitation, and the risk of complications, such as adhesions. These may have been cases in which the locations of snapping were similar to other snapping popliteal tendon cases but the pathogenesis was slightly different. In our case report with a younger patient, there was no osteophyte on plain radiography or MRI. It is significant to mention that the condition could be treated through tubercle excision and debulking of hypertrophied tissue with arthroscopy alone even in a case with a young patient without a more protruded osteophyte.In the case of tubercle excision, some cases required open excision, but excision with a sufficient margin could be made with all arthroscopic procedures alone, as was done in the present study. In 2 cases, Gain et al also showed that it could be treated through arthroscopic osteophyte excision. However, in the cases of Gain et al, the mean age of the patients was slightly higher, and the condition was not associated with the athletic lifestyle. A clear osteophyte was visible on both plain radiography and arthroscopic examination.It is difficult to determine whether popliteal tendon release or debulking, tenodesis with or without sulcus deepening, or tubercle excision is the best method for treatment, as only a few cases have been reported until now. However, if this condition can be treated with a less invasive treatment, the less invasive method should be performed.Snapping popliteus tendon syndrome is a disease that is hardly recognized due to its low prevalence and difficulty in diagnosis, and it requires close observation of the patient before surgery. The location of the tenderness and the snapping occurrence must also be carefully identified. Based on this, it is important to accurately diagnose and resolve the pathology of popliteus tendon without any misdiagnosis. Our procedure is an entirely arthroscopic technique; as it has the prominent advantage of a speedy recovery and easy rehabilitation, it could also be helpful to set treatment standards for this disease in the future.Conceptualization: Dong Hwan Lee.Formal analysis: Ji Hyun Ryu.Methodology: Se-Won Lee, Dong Hwan Lee.Supervision: Se-Won Lee, Dong Hwan Lee.Writing – original draft: Ji Hyun Ryu.Writing – review & editing: Dong Hwan Lee.


Dear EditorWe present the clinical case of a 41-year-old woman with a medical history of iron deficiency anemia. The patient came to the consultation for abdominal pain, located in the epigastrium, and an alteration of the intestinal rhythm of 9 months evolution, referring to tenesmus and occasional hematochezia. An analysis was carried out ; low ferritin (4.8 ng/mL) was observed, with the rest of the study normal. To complete the study, abdominal ultrasound, gastroscopy, and colonoscopy were requested.The gastroscopy revealed 2 hairy lesions in the esophagus 20-25 cm from the dental arch: these lesions were 2-3 mm in diameter each and had an appearance suggestive of papillomas . These w1Esophageal papilloma is an infrequent entity, with an approximate incidence that ranges between 0.01% and 0.04% in the population. They are seen more frequently in patients with tylosis and acanthosis nigricans, and there is an association with Goltz syndrome, a congenital disorder of focal dermal hypoplasia characterized by hyperpigmentation, sclerodactyly, dysplastic changes in teeth and bones, and papillomas in the perianal, oroesophageal, and genital regions.2 The second theory relates these lesions to HPV infection. Studies carried out in populations of Italy and Hungary have documented the existence of HPV in 26-41% of esophageal papillomas.3 However, other studies in populations of the United States, Finland, and Poland have identified HPV in less than 5% of cases.4 The role of HPV in pathogenesis is supported by the fact that the most frequently found serotypes are 6 and 11 . The route of acquisition of this viral infection is most often oral sex, but it has been considered possible via the ingestion of cellular particles from warty lesions from the hands and genitals.5 The evolution of these lesions to malignant forms is not frequent, although in cases with other concomitant risk factors, this evolution can be seen.Pathogenesis is uncertain, although there are 2 proposed theories: the first is the inflammatory theory. Approximately 70% of papillomas occur in the distal third of the esophagus and have been associated with reflux, esophagitis, or irritants of the mucosa.10 as seen in the image of the case study described in Diagnosis is usually by chance finding since they are mostly asymptomatic, but long-standing lesions can cause dysphagia if they are of considerable size. The macroscopic appearance on endoscopy is of small, whitish-pink exophytic projections, similar to warts, as seen in


KIT or PDGFRa mutations on molecular analysis. Moreover, we review the pertinent literature. The mesentery constitutes a common location for the metastatic spread of malignant gastrointestinal tumors. Primary mesenteric tumors, on the other hand, are very rare; lymphomas are the most common, followed by benign and malignant mesenchymal tumors. We present a case of a 43-year-old patient operated on for a primary mesenteric leiomyosarcoma with a positive immunostain for DOG1, despite having no  Leiomyosarcoma (LMS) is a malignant mesenchymal tumor originating from smooth muscle cells or from their precursor mesenchymal stem cells . It usuaThis article was presented as a meeting abstract at the 33rd European Congress of Pathology (ECP 2021) on August 29-31, 2021.A 43-year-old patient was admitted in March 2020 due to a sizeable mesenteric mass discovered during a previous myomectomy for uterine leiomyomas (performed elsewhere) in October 2019. History was significant for cervical conization due to high-grade squamous intraepithelial lesion (HGSIL) fibroids. On laboratory investigation, inflammatory response markers and cancer tumor markers were within normal limits. The patient underwent a magnetic resonance imaging that revealed a mass with dimensions 4.7x4x3.3 cm that probably originated from the bowel wall or its mesentery with intermediate signal intensity in sequences T1 and T2 and intense inhomogeneous contrast enhancement. The patient was discussed in our local multidisciplinary tumor board (MDT), and the decision was made to proceed with surgical excision of the lesion. We performed an open excision of a mass that arose from the mesentery of the ileum. The tumor was excised with wide margins; it was of whitish color and firm texture. On microscopic examination, the tumor was seen to consist of fascicles of moderately pleomorphic, atypical spindle cells Figure  with up KIT or PDGFRA genes. Based on the above findings, a diagnosis of malignant mesenchymal tumor consistent with low-grade leiomyosarcoma of the mesentery was rendered. A review of slides from the previous myomectomy specimens confirmed the diagnosis of leiomyomas.Molecular analysis  disclosed no mutations of A mass within the transversus abdominis muscle was found four months later during a postoperative check-up. Microscopic examination of the surgical specimen disclosed long, sweeping fascicles of uniform spindle cells with minimal cytologic atypia and low mitotic activity (one mitosis per 10 high-power field (HPF)) set within a collagenous stroma Figure , 2B. TumA diagnosis of abdominal fibromatosis/desmoid was therefore rendered. The MDT decided not to administer adjuvant therapy. At the two-year follow-up since surgical excision, the patient is free from any recurrence of either tumor or metastatic leiomyosarcoma.In contrast to metastases from gastrointestinal tumors, primary mesenteric tumors are rare, lymphomas being the most common, followed by usually benign mesenchymal tumors . MesenteIn seventeen cases, follow-up was available, ranging from 3 to 59 months median, 21 months). Eight patients died during the follow-up period, usually due to local recurrence or metastasis to the liver and lung. Two patients were alive with disease, and seven were alive with no evidence of disease. The demographic and clinical data are summarized in Table  months. The management of LMS of the mesentery depends largely on preoperative imaging . On CT, In our case, we used an immunohistochemical panel consisting of SMA, desmin, CD117, and DOG1 to confirm the diagnosis. SMA and desmin staining are positive in almost all smooth muscle tumors compared to 31% and 4% of GISTs . Both CDKIT or PDGFRa mutations, thus favoring the diagnosis of mesenteric LMS since only a small fraction (10-15%) of GISTs are devoid of these mutations [However, DOG1 expression has not been previously reported in a mesenteric LMS. In our case, uniform positive staining for SMA, desmin, and DOG1 was confusing. Despite the lack of CD117 immunopositivity, a GIST diagnosis could not be ruled out since DOG1 is a more sensitive marker than CD117 . We, theutations .Regarding prognosis, LMS of the mesentery has an aggressive clinical course with an unfavorable prognosis . The oveTumor recurrence can occur within five years in approximately 50% of patients despite adequate local control . CarefulThere is no clear precedent for treating mesenteric LMS, probably due to the small number of cases reported . TreatmeMesenteric LMS is an infrequent entity. Its diagnosis requires a combination of appropriate morphology and immunohistochemistry. As evidenced by the case we present, molecular analysis is necessary to make the correct diagnosis when morphological and immunohistochemical findings are inconclusive.


Acute graft versus host disease (GVHD) is a serious complication of allogenic haematopoietic cell transplantation. The standard treatment of acute GVHD is immunosuppressive drugs, but treating patients who do not respond to these drugs remains particularly challenging. Herein, we report a case of severe acute intestinal GVHD that caused multiple strictures of the small intestine, warranting surgical resection.A 13‐year‐old boy with myelodysplastic syndrome underwent bone marrow transplantation (BMT) from an HLA‐matched unrelated donor. At 38 days post‐BMT, he presented with diarrhoea and abdominal pain, and acute intestinal GVHD was diagnosed.Since his GVHD was steroid resistant, anti‐thymocyte globulin, methotrexate, and mesenchymal stromal cells were administered. His symptoms gradually improved over 6 months post‐BMT. However, 8 months post‐BMT, severe abdominal pain recurred. Multiple strictures of the ileum were observed on contrast‐enhanced X‐ray imaging and computed tomography (top); these strictures were assumed to be the fibrostenotic consequence of severe acute GVHD. These multiple strictures were so severe that passage of soft foods could not be expected. Ten months post‐BMT, he underwent temporary ileostomy to circumvent the strictures, and abdominal pain was ameliorated. Fifteen months post‐BMT, he underwent resection of the constricted ileum and ileostomy closure. Multiple strictures of the ileum were confirmed (bottom), and pathological inspection revealed fibrosis of the mucosa with no signs of persistent GVHD. The patient is now free from any gastrointestinal symptoms.Severe acute intestinal GVHD induces strong inflammation and could lead to multiple severe fibrostenotic strictures of the intestine. Because acute GVHD is a systemic disease, surgery is not generally considered. The present case highlights the importance of recognising that a small subset of patients undergoing stem cell transplantation might develop severe intestinal GVHD warranting surgical intervention.Hisashi Ishida wrote the manuscript. Hisashi Ishida, Takuo Noda, Seiji Kawano, Maho Sato, Hirokazu Tsukahara, and Akira Shimada participated in patient care. All authors reviewed the manuscript.The authors declare no conflict of interest.Written informed consent was obtained from the patient and his mother.