Schwannomas are benign tumors that often occur in the medulla spinalis. It arises from the Schwann cells which form the sheath of peripheral nerves. Schwannomas comprise about 30% of primary intraspinal neoplasms and approximately 75% in intradural, 10% intra-extradural, and 15% extradural. Purely extradural schwannoma is rare.We present two adult males with extradural thoracal schwannoma. In this case, the main complaint is paraparesis and followed by suffered low back pain and spastic. The initial symptoms of this tumor depend on the level of the tumor location. Usually begins with localized pain, sharp and transient, followed by radicular pain and radiculopathy. We reported cases of schwannoma of the thoracal spine that have presented with neurological involvement. As a result of contrast, an MRI of the spine showed tumor extradural mass lesion extending from thoracal 1–2 vertebral and thoracal 4–6 vertebral level.The location of this tumor is rare because located in the extradural (15%), which are present between the bone structure and the dura. Surgery is the treatment of choice, in this case, usually results from excellent prognostic. This patient underwent hemilaminectomy and complete surgical resection.Early diagnostic and complete surgical resection before the occurrence of severe symptoms will show an excellent prognosis. •Purely extradural schwannoma is rare.•We presented a schwannoma of the thoracal spine that have presented with neurological involvement.•Early diagnostic and complete surgical resection before the occurrence of severe symptoms will show an excellent prognosis. It aris2We report 2 cases of patients with extradural thoracal schwannoma.2.1The first case is 60 years old male who presented to us with paraparesis and spastic for more than 3 months. The patient also had low back pain. He had no abnormality of bladder dan bowel habits. His past medical history and family history were insignificant. On examination of the lower extremity, there was paraparesis with a power of 3/5, increased physiological reflex, and positive Babinski pathological reflex. No sensory level defect was detected and no sign of spinal deformity.His laboratory and thorax X-ray showed no apparent abnormalities as well. Magnetic Resonance Imaging (MRI) revealed findings of benign soft tissue consisting of proliferating cells with a spindle shape partly with hypercellular areas and partly with hypocellular areas. The conclusion was that the tissue was a schwannoma tumor. .Fig. 1Ma2.2A Male 20 years old was admitted to the neurosurgery department with a main complaint of paraparesis 6 months ago. The patient also suffered low back pain and spastic. He had no abnormality of bladder dan bowel habits. His past medical history and family history were insignificant. His physical examination was normal. Neurological examination reveals paraparesis with the power of 2/5 in both lower limbs, hypertonia in bilateral lower limbs, and increased tendon reflexes in the lower limbs. Babinski reflexes were positive in bilateral lower limbs. No sensory level defect was detected and no sign of spinal deformity.His laboratory and thorax X-ray showed no apparent abnormalities as well. In imaging contrast, an MRI of the spine showed tumor extradural mass lesion extending from thoracal 1–2 vertebral level . The patWe diagnose the patient from clinical findings and imaging methods and MRI is the best method for diagnosis and differential diagnosis. The size and specific margins of the mass demonstrate the localization and invasion of the contiguous structures. We treat all patients with hemilaminectomy and complete surgical resection, after follow-up 3 months after surgery, the patient becomes normal from paraparesis and spastic .Fig. 4EvIn these two patients, we only performed a hemilaminectomy and excision of the tumor with a guiding microscope, we did not install a pedicle screw because it was unnecessary and too excessive in our opinion, in principle, according to the surgical procedure, we could reach the tumor without destroying the stability of the spine. in both patients we used a guiding microscope without IOM (intra operative monitoring), our center is rural area, we don't have IOM but we try to give the best for patients.3Schwannoma is a common nerve sheath tumor that usually arises from sensory nerves and is benign . Some clAlqroom, R. et al. the most common initial symptom was radicular pain (67%) followed by motor weakness, another 31% had myelopathy and sphincter disturbances [In this case, the main complaint is paraparesis and followed by suffered low back pain and spastic. The initial symptoms of this tumor depend on the level of the tumor location. Usually begins with localized pain, sharp and transient, followed by radicular pain and radiculopathy. When the compression of the tumor in the spinal cord increases, will lead to damage to the spinal cord and causes myelopathy . Numbnesurbances . Some sturbances .In some cases, the location of schwannoma was more common in the cervical region  and lumbar than thoracal . In the We reported cases of schwannoma of the thoracal spine that have presented with neurological involvement. MRI of the spine showed tumor extradural mass lesion extending from thoracal 1–2 vertebral and thoracal 4–6 vertebral level. The location of this tumor is rare because located in the extradural (15%), which are present between the bone structure and the dura. Magnetic resonance imaging (MRI) is the gold standard to diagnose especially in spinal tumors . In MRI,Sridhar et al. suggested a classification of benign Spinal Schwannoma in various types. Type I-intraspinal tumor, <2 vertebral segmented in length; a:intradural; b: extradural. Type II-intraspinal tumor, >2 vertebral segments in length (giant tumor). Type III-intraspinal tumor with extension into nerve root foramen. Type IV-intraspinal tumor with extraspinal extension (dumbbell tumors); a: extraspinal component <2.5 cm; b: extraspinal component >2.5 cm (giant tumor). Type V-tumor with erosion into vertebral bodies (giant invasive tumor), lateral and posterior extensions into myofascial planes [Park et al. modified the classification by adding two new classifications. Type VI-tumor in an entirely intravertebral location without intraspinal portion. And type VI-intraspinal tumor with erosion into vertebral bodies and extension into nerve root foramen [According to l planes . And Par foramen . TherefoOn histopathological examination, showed polymorphic spindle cells with area hypercellular and hypocellular, without malignancy sign. Spinal schwannoma has two types in histopathological findings, Antoni A (hypercellular) and Antoni B patterns (hypocellular) ,30. AccoSurgery is the treatment of choice in this case, which usually result from excellent prognostic . This pa4Spinal extradural schwannoma is a rare case, especially in thoracal. Early diagnostic and complete surgical resection should be the goal treatment, these also will significantly show good prognostic.Obtained.No funding or sponsorship.RHA, BAM, NUW, WYD, and KVG wrote the abstract, introduction, case, discussion, conclusion. RHA, BAM, NUW, WYD, and KVG performed critical edits and final revision, figures.1.Name of the registry: NA.2.Unique Identifying number or registration ID: NA.3.Hyperlink to your specific registration (must be publicly accessible and will be checked): NA.Rohadi Muhammad Rosyidi.The authors report no conflict of interest concerning the materials or methods used in this study or findings specified in this paper.Not commissioned, externally peer-reviewed.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.None.


Pheochromocytoma of the Adrenal gland Scaled Score (PASS) 2. The evolution was favorable with normalization of blood pressure and urinary catecholamines at one week, three months, six months, and one year. He tested negative for hereditary syndromic including Von Hippel-Lindau (VHL), RET genes, subject to the succinate dehydrogenase complex B and D subunit genes (SDHB-D), which have been requested.Pheochromocytomas and paragangliomas are rare neuroendocrine tumors developed from chromaffin cells. They are exceptional in children with an atypical symptomatology. We report here a case of a 17-year-old boy who presented with a left retroperitoneal mass discovered on thoracic-abdominal-pelvic computed tomography (CT) scan in the presence of diffuse abdominal pain, more pronounced in the left hypochondrium. The exploration had objectified an exclusive secretion of urinary normetanephrine over 24 hours. Metanephrine and 3 ortho methyldopamine were within normal limits. He had adrenalectomy after controlling his blood pressure with an alpha blocker. Histology had confirmed a pheochromocytoma of non-aggressive potential  Pheochromocytomas and paragangliomas are rare tumors that develop from chromaffin cells, producing an excess of catecholamines . Their mThere is a variation in catecholamine secretory phenotype between pheochromocytomas and paragangliomas, with primary or exclusive normetanephrine secretion in paraganglioma, whereas metanephrine secretion is usually confined to pheochromocytomas originating from the adrenal medulla .Here we report a case of pheochromocytoma in a 17-year-old adolescent with excess normetanephrine but normal metanephrine levels.A 17-year-old adolescent presented with an incidentally discovered left retroperitoneal mass measuring 6 × 7.2 × 7.5 cm on a thoracic-abdominal-pelvic computed tomography (CT) scan performed in the face of diffuse abdominal pain that was more pronounced in the left hypochondrium . Metanephrine and 3 ortho methyldopamine in 24-hour urine were within normal limits. The workup for extension and secondary localization was unremarkable.The patient benefited during 10 days from a preparation to the surgery by an alpha blocker prazosin to control his arterial pressure, introduction of a calcic inhibitor after 7 days, and a good hydration to increase the blood volume and avoid postsurgical hypotension. A preoperative embolization of the superior and inferior adrenal arteries was performed. The procedure was uneventful.The patient underwent a left adrenalectomy, with the notion of three adrenergic crisis during the surgical procedure. The post-surgery course was uneventful.His blood pressure was stable after the surgery. Histology had confirmed the diagnosis of pheochromocytoma of non-aggressive potential (PASS score 2). The evolution was favorable with normalization of blood pressure and urinary methoxylated derivatives at one week, three months, six months, and one year after surgery Table .As part of the genetic syndromic workup, the search for genetic mutations including Von Hippel-Lindau (VHL) and RET genes were negative; the B and D genes of the succinate dehydrogenase complex (SDHB-D) are still in progress.Pheochromocytomas and paragangliomas are rare neuroendocrine tumors. Although widely described in the adult literature, in the pediatric population pheochromocytomas remain less frequent and poorly described .The clinical presentation is atypical. Excess of secreted catecholamines lead to hypertension, excessive sweating, and tachycardia. Other symptoms may be present, such as abdominal pain.Paragangliomas primarily secrete normetanephrine, whereas metanephrine secretion is usually limited to pheochromocytomas arising from the adrenal medulla . One hypOur patient had an adrenal pheochromocytoma producing exclusively normetanephrine. One study found that noradrenergic tumors generally exhibit continuous hypertension, and metanephrine-producing tumors cause paroxysmal hypertension , which iMore than 40% of affected patients have identifiable germline mutations; therefore, international guidelines suggest genetic testing for all patients regardless of age -7. The mSurgical resection is the treatment of choice, along with pre-operative preparation to prevent cardiovascular complications by alpha-blockers, calcium channel blocker, and good hydration to prevent severe hypotension after tumor removal . Open suPheochromocytoma and paraganglioma are rare tumors in the pediatric population. Their diagnosis is suspected in the presence of an adrenal mass or hypertension and is based on the determination of urinary catecholamines. Paragangliomas primarily secrete normetanephrine, whereas metanephrine secretion is usually limited to pheochromocytomas. Our case report shows an adrenal pheochromocytoma producing exclusively normetanephrine. The management is medical-surgical and multidisciplinary. Radical resection is the treatment of choice. Genetic testing is important in patients with pheochromocytoma/paraganglioma.


Proboscis lateralis (PL) is an uncommon congenital facial deformity marked by the protrusion of a primitive tubular structure made up of skin and soft tissue that generally emerges from the eye's medial canthus and is associated with some craniofacial deformities. We report the first case of PL with multiple craniofacial, neurological, cardiac, and spinal anomalies.A full-term female baby delivered by cesarean section cried immediately at birth. The mother reported having a normal pregnancy but has a history of x-ray during her first trimester. The baby was born with a rare presentation of proboscis lateralis which was accompanied by multiple anomalies, including but not limited to bilateral colpocephaly, corpus callosum agenesis, complex cyanotic congenital heart disease, and hemivertebra of the T10 body.PL is an uncommon congenital condition that causes a variety of craniofacial abnormalities. Multiple additional defects affecting various organ systems should also be evaluated in a person diagnosed with PL. Proboscis lateralis (PL), first described by Forster in 1861, is a rare congenital facial anomaly usually due to incomplete nose formation in utero. This abnormality is characterized by a primitive tubular structure composed of skin and soft tissue protruding from the facial surface. The medial canthus of the eye is the most commonly reported attachment site of PL; However, rarely has the PL been found attached to other regions such as the lateral canthus, lateral supraorbital ridge, mid-upper eyelid, midline of the root of the nose, and the chin . PL is uA five-day-old full-term female baby delivered by cesarean section cried immediately at birth. The mother had no comorbid conditions and described her pregnancy as normal. During the pregnancy, the mother had an X-ray of her leg, but she was unaware of her pregnancy at the time. There is no family history of congenital anomalies, and the baby is born to parents that are unrelated. The baby was born with a trunk-like appendage that was arising from the superomedial canthus of the left eye, 26 mm × 12 mm in size, with a small tract with Cerebrospinal fluid discharge. The left nasal cavity was hypoplastic with left anophthalmia. No cleft palate, cleft lip, or choanal atresia was noted.  revealed complex cyanotic congenital heart disease, including double outlet Right Ventricle with a large ventricular septal defect of 8 mm, malposed great vessels, mild valvular right ventricular outflow tract obstruction (RVOTO) with a pressure gradient of 26 mmHg, small Patent ductus arteriosus (PDA) with a left to right shunt and confluent branched pulmonary arteries. Two ostium Secundum atrial septal defects (ASD) were noted of 4 mm and 25 mm with a normal biventricular structure and function. On Magnetic resonance imaging (MRI)  [In 1985, the Boo-Chai classification was developed, taking different possible features of proboscis and the other associated defects into account. The categorization split cases of PL into 4 groups. Group-1 is described as a person with proboscis but a normal nose, observed in 9% of the cases studied by Boo-Chai. Group-2 is proboscis, with nasal defects seen in 23% of the included population. Group-3 is taken as proboscis with a nasal defect along with defects of either the eye or ocular adnexa, seen in 47% of cases. Group-4 has all the features of group-3 with the addition of a cleft lip or palate, which was found in 21% of the cases in the research [Later Sakamoto et al. added the component of intraorbital distance to the grouping of PL done by Boo-Chai, along with an addition of two more groups. Group 5 is defined as a PL with hypertelorism with encephalocele, a nasal, eye, and ocular adnexa defect, with either cleft lip or palate and Cerebro-oculo-nasal syndrome. Group 6 is similar to group 5 but presents with hypertelorism without encephalocele and, instead of Cerebro-oculo-nasal syndrome, presents with Holoprosencephaly .Common CNS abnormalities seen with PL are encephalocele and Holoprosencephaly, but cases with other CNS abnormalities like meningoencephalocele, hydrocephalus, sphenoorbital basal cephalocele, corpus colosseum agenesis, arachnoid cyst, and brain stem asymmetry have been reported , 11–16. The spinal deformity was reported only in one other case, where the patient had a malformation of the cervical vertebra along with PL . Our casThe mother's exposure to radiation during the X-ray in the first trimester could be a contributing factor to the extensive list of abnormalities in the child. However, single diagnostic scans are considered safe by both the American College of Obstetricians and Gynecologists and the American College of Radiology. A single lower limb X-ray exposes the fetus to 0.0001 rad of radiation, whereas a minimum dose of 5 rad is estimated to be potent enough to cause congenital abnormalities , 19.The management of PL requires A multidisciplinary approach involving several departments such as pediatrics, otorhinolaryngology, plastic surgery, anesthesiology, ophthalmology, radiology, and psychiatry services is essential for successfully managing functional and aesthetic aspects of PL. Cases involving the cardiovascular and nervous  systems, like the one we reported, required additional opinions from respective departments. The treatment of PL has long been a matter of debate. There is still no consensus about the time when treatment should start but early management in childhood is recommended to mitigate the psycho-social consequences of PL .PL is a rare congenital disorder associated with multiple anomalies. A person diagnosed with PL should be checked for multiple other anomalies involving other organ systems, even the ones not commonly associated with PL. As our patient had multiple anomalies like bilateral colpocephaly, hemivertebra of the T10 vertebral body, and multiple cardiac anomalies, which have previously never been reported in the literature. In addition, due to the effect of PL on the patient’s quality of life. Treatment should aim to achieve an acceptable appearance to ensure the patient’s psychological wellbeing and allow healthy social interactions. Parents need to be counseled to encourage early intervention in children born with PL.


Anomalous systemic arterial supply to the normal basal segment of the left lower lobe is a congenital abnormality of the lung, frequently and is generally diagnosed at a young age. Surgery is generally recommended if symptoms such as blood sputum or fever are observed. Resection of the abnormal artery is often performed at an early age, with only few reports of surgery being performed at an older age. In addition, to the best of our knowledge, there are no reports on surgical treatment of abnormal calcified vessels to date. Herein, we have presented a case in which a calcified aberrant vessel of lung was resolved surgically.A 65-year-old female, previously diagnosed with anomalous systemic arterial supply to the left normal basal segment of the left lung lower lobe of lung was under observation on the basis of being asymptomatic.The patient presented to the emergency room with the chief complaint of blood in the sputum and she was referred to our hospital for a surgery. Computed tomography showed circumferential calcification of the intima of the abnormal vessel, which might have contributed to incomplete resection of the artery if automatic sutures were used. Thus, the abnormal vessel was ligated and dissected using pledgeted 4–0 polypropylene sutures and vessel clips under open thoracotomy followed by left lower lobectomy. The patient was discharged seven days after surgery without any serious surgical complications.Vascular congenital anomalies of the lung are often operated at a young age presenting due to the associated symptoms. However, even if the disease is discovered incidentally and does not cause any symptoms or calcification in the aberrant artery, early surgical intervention is important due to the possibility of calcification occuring in the future. This can help minimize the degree of surgical invasion. Anomalous systemic arterial supply to the normal basal segment of the left lower lobe of the lung is a rare congenital abnormality. This abnormality had previously been classified as a type of pulmonary sequestration (PS) by Pryce . HoweverSurgical treatment has been reported for the management of this rare congenital abnormality. The surgical approaches that can be employed vary widely, and can include ligation, lobectomy, segmentectomy, and coiling, performed either via thoracoscopy or open thoracotomy –10. OnlyHerein, we report a case of a calcified abnormal vessel of the lung, safely using reinforced ligation.A 65-year-old woman was diagnosed with anomalous systemic arterial supply to the normal basal segment of the lung in her early forties. As she was asymptomatic, she had been under observation only since the time of her diagnosis. Her medical history included hypertension; she was found to have hypertension two years ago when she was admitted to a previous medical emergency for hypertension.She reported to the emergency room of her previous doctor with the chief complaint of blood in her sputum. After a computed tomography (CT) scan, the anomalous systemic arterial supply to the normal basal segment was suspected as the cause of the presenting symptom, and surgery was considered necessary. Thereafter, the patient was referred to our hospital for surgical treatment.CT images revealed a calcified aberrant artery with maximum diameter of 11 mm at the root. The vessel originated from the descending aorta at the level of the eighth to ninth dorsal ribs Figs. . ThickenPulmonary function examination showed a forced expiratory volume per second of 1.69L (80.1% of the predicted volume) and a forced vital capacity of 2.11L (77% of the predicted volume). Echocardiography showed good cardiac contractility with an ejection fraction of 65%. The patient’s TR max was 2.4 m/s, TAPSE was 10.2 mm, and TRPG was 23.2 mmHg.We performed a left lower lobectomy with aberrant artery excision. First, the patient was placed in the left lateral recumbent position under general anesthesia Fig. . The thoHistopathological examination of the surgical specimen revealed elastic type artery exhibiting intimal thickening and lumen dilation with calcification. On its peripheral side, some atheroma formation was also observed.Anomalous systemic arterial supply to the left basal lung is a relatively rare disease. This pathology was classified by Pryce et al. as type I sequestration, with no abnormalities in the bronchial branches .Patients with this anomaly often need surgery because of the risk of hemoptysis due to pulmonary hypertension associated with left-to-left shunt . SurgeryIn the past, we had resected aberrant arteries of this disease using autosutures under video-assisted thoracoscopic surgery (VATS) in all cases presented with this disease. However, in the present case, we decided to handle the aberrant artery using ligation with clips under thoracotomy, based on the concern that incomplete dissection by automatic suture machine due to the dense calcification could lead to uncontrollable bleeding from the dissected end due to the dense calcification. Hence we rigorously dissected the vessel using two 4–0 polypropylene sutures and two vessel clips and were able to complete the surgery without bleeding.Another option in such cases is to treat the vessel on the peripheral side of the calcification with an automatic suture. However, there is a risk of aneurysm formation in the sutured area due to pressure from the left ventricular system. The firIt seemed to occur over time with advancing age under the condition of the left ventricular system. The only case of calcification of an aberrant artery reported so far occurred in a 53 year old patient. The lack of other reports of similar calcification can be attributed to the fact that surgery is often performed for these patients at a young age in these pationts.Considering the above, it can be inferred that in asymptomatic cases with accidental detection of anomalous arterial supply, patients are likely to be kept on regular follow up. In the present case, however, the calcification of the abnormal blood vessel forced us to perform open thoracotomy to treat the vessel. Calcification of abnormal blood vessels can complicate VATS and necessitate invasive surgery. Therefore, even if there are no symptoms at the time of diagnosis, given the possibility of calcification occurring in the future, surgical options should still be considered at the time of diagnosis.The present paper describes only one case; it is therefore necessary to evaluate a larger number of cases. In addition, the reported patient’s previous CTs were performed at other hospitals and could not be obtained this time. Thus, it was not possible to assess the course of changes in the calcified lesions, and it remains debatable whether abnormal vessels do indeed calcify with age.Early surgery is recommended for the management of anomalous systemic arterial supply of the lung, considering that calcification of abnormal blood vessels may occur with age, requiring more invasive procedures for treatment in the future.


The present case contributes to the limited literature on delayed chest wall hematomas following blunt trauma. The literature review provides a summary of similar previously reported cases.We report the case of a 59-year-old Caucasian male who presented to the emergency department with a rapidly expanding chest wall hematoma. Six weeks earlier, he had sustained multiple rib fractures and a pneumothorax after falling 4 m from a ladder. Computed tomography angiography was used to identify two sources of active bleeding on the left dorsal scapular artery. The patient underwent surgery with evacuation of the hematoma and ligation of the artery. The patient was hospitalized for 3 days and recovered with no sequelae.A literature review revealed eight previously reported cases of chest wall hematomas exterior to the endothoracic fascia following blunt trauma. Most cases were initially diagnosed by computed tomography of the chest and finally by angiogram. Management options range from surgical drainage to angiographic embolization. This case is unusual regarding the delay in the development of the hematoma and illustrates the importance of considering this diagnosis even weeks after relevant trauma. Blunt injuries account for the majority of chest traumas and cause a range of complications, including rib fractures, pneumothorax, hemothorax, and chest wall hematomas . ArteriaA 59-year-old Caucasian male was admitted to the emergency department with a rapidly expanding mass in the thoracic wall in relation to the inferior part of the left shoulder blade. His medical history included mild renal insufficiency and epilepsy. He did not take any antiplatelet or anticoagulant medication. Six weeks earlier he fell 4 m from a ladder, resulting in multiple rib fractures and a left-sided pneumothorax diagnosed on chest radiography. The pneumothorax was treated with a chest drain placed in the left anterior axillary line at the level of the papilla. Pain-relieving medication included paracetamol, ibuprofen, and oxycodone, and the patient was released from the hospital after 5 days of treatment.The mass in the thoracic wall had appeared the previous evening about 8 hours before the patient came to the hospital and kept expanding slowly during his stay in the emergency department. He had pain in his left shoulder, and his left-hand fingers felt cold and tingling with no further neurological symptoms. A clinical examination revealed a skin-colored, sore swelling approximately 10 cm in diameter in relation to the inferior angle of his left shoulder blade. Vital parameters were normal. Blood results showed hemoglobin levels of 9.18 g/dL compared with 11.92 g/dL 6 weeks earlier.Thoracic computed tomography angiography (CTA) revealed a large hematoma in relation to the left scapula located between the costae/intercostal muscles and the serratus anterior muscle. The hematoma measured 7 cm × 14 cm × 18 cm  equaling a volume of approximately 930 mL Eq. .The patient remained stable and was transferred to the department of cardiothoracic surgery. Open exploration was chosen because of compression symptoms. At primary surgery, the hematoma was evacuated beneath the left latissimus dorsi. The bleeding source was not found, and the resulting cavity was packed with gauze binding. At second look the following day, the scapula was lifted, coagulated blood was removed, and the bleeding sources on the left dorsal scapular artery were identified and ligated. The patient was discharged from the hospital after a total of 3 days. Six months later, the patient was well with no sequelae.We conducted a review of the literature to identify how previous cases of chest wall hematomas exterior to the endothoracic fascia following blunt trauma have been diagnosed and managed. We hypothesized that most cases were diagnosed by CTA of the chest and managed by surgical drainage similarly to our case.The literature search was conducted in PubMed to identify eligible articles. The search was divided into four main categories using MeSH term searches. The four categories were (1) “Thorax,” (2) “Blunt trauma,” (3) “Hemorrhage,” and (4) “Arterial bleeding.” MeSH terms within one of the four categories were combined using Boolean operators “OR.” Finally, all four categories were combined using the Boolean operator “AND.” Two authors  conducted the search individually to secure validation of the method. The MeSH term search was followed by an “All Fields” search in PubMed to expand the search. Lastly our own reference list was screened.MeSH search string  AND  AND (“Blood”[MeSH Terms] OR “Hemorrhage”[MeSH Terms] OR “Hematoma”[MeSH Terms]) AND (“Thoracic Arteries”[MeSH Terms] OR “Computed Tomography Angiography”[MeSH Terms] OR “Aneurysm”[MeSH Terms])All fields search string (scapular artery) AND (blunt trauma)Inclusion criteria: (a) articles in English, (b) articles published after year 2001 , (c) hematomas exterior to the endothoracic fascia, (d) mechanism: blunt trauma, (e) confirmed diagnosis. Exclusion criteria: (a) hemothorax, (b) cardiac tamponade, (c) unknown vessel, (d) concomitant hematomas interior to the endothoracic fascia, (e) concomitant penetrating trauma, (f) cases in children (< 18 years of age).All papers retrieved from the database were screened by title and abstract by two authors . Then, full texts of relevant articles were screened by two authors  to identify the papers that satisfied the eligibility criteria for the review. The identified publications references were hand-checked to find additional studies. Studies were then excluded or kept on the basis of their consistency with the inclusion and exclusion criteria. The PRISMA flow diagram was used to record the selection process Fig. .Fig. 1PRInformation was retrieved on mode of diagnosis, which artery was damaged, whether it was in relation to any fractures, and how the hematoma was managed Table . InformaAs seen from the PRISMA flow chart Fig. , the seaThe results are presented in Table All cases were initially diagnosed with a chest computed tomography (CT), two of those with a contrast-enhanced scan. Seven out of eight cases were finally diagnosed with an angiogram, and the last with a contrast-enchanted chest CT. Seven out of eight cases were treated with angiographic embolization of the artery; one of these underwent subsequent surgical drainage. The last case was removed surgically through a limited subaxillary incision.This case illustrates a common complication following blunt thoracic trauma in terms of a chest wall hematoma  but withThere are two possible explanations for the time delay in this case: initial trauma caused two pseudoaneurysms on the dorsal scapular artery ; alternaA CT scan is a sensitive diagnostic tool for hemothorax, pneumothorax, pulmonary contusions, and chest wall hematomas as well as identifying sources of bleeding . This caIn terms of treating a chest wall hematoma, angiographic embolization is an effective and minimally invasive procedure and is commonly performed to stop bleeding from a variety of vessels . HoweverDelayed hematoma from a chest wall artery is a rare complication to blunt thoracic trauma. We present a case where a hematoma of approximately 0.93 L accumulated rapidly 6 weeks after initial trauma due to left dorsal scapular artery bleeding. A review of the literature revealed eight similar cases, five of which were due to pseudoaneurysms and three due to vessel rupture. A contrast chest CT is a useful diagnostic tool for chest wall hematomas, whereas CTA can be used to diagnose hematomas as well as the bleeding sources. The patients should be treated either at a department of cardiothoracic surgery with open exploration or by an interventional radiologist with endovascular embolization .In conclusion, this case report contributes to the relatively limited literature on delayed chest wall hematomas and highlights the importance of considering this diagnosis even weeks after relevant trauma. The systematic literature search provides an overview of diagnostic and management options of chest wall hematomas exterior to the endothoracic fascia.The blood volume was calculated as an ellipsoid: The dorsal scapular artery supplies the levator scapulae and rhomboid muscles and is part of the scapular arterial anastomosis. Most frequently, it branches from the second or third part of the subclavian artery, rarer from the transverse cervical artery.Thoracic imaging.A A 3D reconstruction of the CTA shows two spots of active arterial bleeding (green arrow) and multiple rib fractures. B A 3D reconstruction of the CTA shows spots of active arterial bleeding (red arrow) within the large subscapular hematoma. In addition, multiple rib fractures are shown. C A maximum-intensity projection (MIP) reconstruction of the CTA shows two spots of active arterial extravasation of contrast medium (active bleeding). D and E The CTA shows two focal spots of active arterial bleeding (contrast medium extravasation). F A radial rib range reconstruction of the CTA shows rib fractures from the 3rd to the 12th left rib and several right-sided rib fractures.


There are some reports of localized amyloidosis in the larynx, an entity that corresponds to one percent of all benign tumors of this region. However, there are only two cases of amyloidosis in the Waldeyer's ring 6, 13, 14. We hereby describe a rare case of amyloidosis in areas not associated with the upper aero-digestive tract: tonsil pillar, rhinopharynx, supraglottis and glottis, without visible continuity of amyloid tissue. We will also discuss post-operative follow up with severe dysphagia. Laryngeal amyloidosis accounts for about 1% of all laryngeal benign tumors, more prevalent in males at a 3:It may manifest as a systemic disease, or limited to certain organs resulting from the extracellular deposits of amyloid protein.There are about 20 different types of amyloid protein; however, despite subtle differences in aminoacid sequences, all of them present similar spatial orientation (β-folded), providing them with the quality of being fibrous, insoluble and resistant to proteolysis.They can be primary disorders , secondary to infectious processes such as tuberculosis, leprosy and osteomyelitis or to chronic inflammatory processes such as rheumatoid arthritis , or, still, secondary to post-hemodialysis such as it happens in amyloidosis caused by β2 microglobuline.There is also classification according to the type of amyloid protein such as: light chain (LC), type A (AA), type β2 microglobuline (Aβ2M), heavy chain (HC), among others.Classifications overlap. For example, LC encompasses the primary idiopathic in which there is a monoclonal increase in γ globuline without apparent cause and it also involves the one that is secondary to multiple myeloma. It has worse prognosis, because it is more often associated with systemic disorders, and among the most severe ones we have the cardiac, rarely AA type. Notice that, although laryngeal amyloidosis is of the LC type, it is usually localized.The following report is a peculiar case thanks to, especially, to two aspects: the rarity and multiplicity of the lesions, and also, the difficulty in treating the severe dysphagia that characterized this case.Female, 55 years old, Caucasian, complaining of floating hoarseness, mild respiratory discomfort and dysphagia, especially for grains, lasting for 8 years and with important worsening in the last 3 months. She brought with her the results from a pathology exam done in another center that suggested amyloidosis. As comorbidities she had systemic arterial hypertension, angina and epigastralgia.Nasofibroscopy exam revealed multiple masses: in the rhinopharynx, posterior tonsil lateral wall, laryngeal face of the epiglottis, ventricular folds, aryepiglottic folds, right vocal fold , 2 and 3The patient was then taken to the operating room and the rhinopharynx mass was partially removed with a cutting forceps in order to avoid a large open wound and later local stenosis. The masses in the posterior tonsillar pillar, in the epiglottis, right ventricular fold, and that in the right vocal fold were all removed by laser. We also carried out a tracheotomy in order to secure the airway.In the 2nd post-operative day (2PO), we observed food residues oozing out of the tracheal cannula. We then decided for a diet through the nasogastric tube (NGT). The patient developed a bronchopneumonia because of aspiration and was started on proper antibiotics. On the 7th PO a new test was carried out and we noticed the oozing of food through the NGT. Since she was breathing normally when the cannula was plugged, we decided to remove the cannula. However, even receiving her food through the NGT, she still aspirated. We carried out a function assessment of her swallowing, a swallowing videoendoscopy (SVE) test with thick and pasty-like liquid food per os, and there still was residue left after swallowing, laryngeal food penetration during swallowing, followed by laryngotracheal aspiration and cough reflex. We diagnosed a severe mechanic oropharyngeal dysphagia, with stiffening of the entire supraglottic structures, compromising the efficacy of laryngeal protection mechanisms during swallowing , causing the laryngotracheal food aspiration. She was referred to a speech therapist for swallowing rehabilitation, with emphasis in supraglottic maneuver exercises , and forced swallowing.The patient was doing well in the post-operative; she started to feed by mouth with the therapist support, swallowing only pasty food, still avoiding liquids and solids. Nonetheless, she presented with acute respiratory failure, and it was necessary to perform an emergency tracheotomy, causing an important worsening in her swallowing, and she returned to food intake through the nasogastric tube. She was refereed to a new SVE one month after the emergency tracheotomy, still feeding through the NGT only. We tested liquid, thick liquid and pasty food, and there was penetration of all of them . She undThe patient followed the instructions properly and really helped out during the sessions, and she improved. A new SVE after one week under subglottic - conventional vision did allow proper visualization of the glottic slit and, consequently, of laryngotracheal aspiration. We did not observe aspiration with any of the food types during the test, although there was contrast spill over the laryngeal vestibule, above the vocal folds. She was diagnosed with moderate oropharyngeal dysphagia, with good compliance with the supraglottic maneuver, and this allowed us to remove the NGT and to keep her feeding per os.In the head and neck, amyloidosis may involve the noseAmyloidosis of the upper aerodigestive tract is usually localizedIt is frequent to have multiple laryngeal sites or, even, that of the aerodigestive tractIt is imperative that the otorhinolaryngologist, when facing a diagnosis of aero-digestive tract amyloidosis, understand the need for systemic investigation. In order to investigate systemic diseases, we need tests that help us assess the function of possible target organs, such as the kidneys, liver, lower respiratory tract, digestive tract, heart and also tests that track down chronic systemic disorders such as multiple myeloma, tuberculosis and rheumatoid arthritis. Fine needle aspiration of the subcutaneous abdominal fat is the exam of choice to detect systemic amyloidosisOur patient had heart symptoms; however, all the tests told us she did not have amyloid heart infiltration, but rather a coronary disorder secondary to chronic systemic hypertension.Our patient complained of dysphonia, dyspnea and dysphagia. She already had had dysphagia for some time; however it worsened in the post-operative, very likely because of post-operative scar fibrosis, with stiffening and changes in the mechanisms that protect the supraglottis and also the tracheostomy cannula that prevents the mechanical movement of effective laryngeal elevation and anteriorization to control dysphagia.In the literature, the most reported symptom of patients with laryngeal amyloidosis is hoarsenessOur case is the 4th report of amyloidosis in the Waldeyer's ring. BeiserDiagnosis is carried out through biopsy in the suspected organ (Congo Red Dye under polarizing light microscopy). It can also be confirmed by the negative potassium permanganate proteolysis test.In the case of symptomatic and localized amyloidosis, as it usually happens in cases of laryngeal amyloidosis, the lesion must be removed and the patient must be started on periodic treatment, because local and distant relapses are commonTreatment is based on surgical resection. As it happened in our case, in some cases, tracheostomy is necessary in order to secure the airwayThe prognosis for laryngeal amyloidosis is excellent if the lesions are completely removed. In certain cases (asymptomatic) the mass can be seen, in other words, clinical follow up.Laryngeal amyloidosis is a differential diagnosis among the lumps and lesions in the upper aero-digestive tract and its location is diverse, in some cases, multiple. We must also bear in mind that the lesion may be a consequence or be associated with some systemic diseases, which must be treated in order to prevent amyloidosis from becoming systemic. Dysphagia is a symptom that, although rare, may happen in amyloidosis patients, often times indicating failure in laryngeal competence against laryngotracheal aspiration.


Objectives: Sialolithiasis is the most common cause of calcifications detected with ultrasound in patients with chronic inflammatory symptoms and swellings of the salivary glands. Other differential diagnoses of calcifications are extremely rare and mostly benign. Methods: Case report and literature review. Results: Two rare cases of malignant parotid gland tumors with calcifications in a localization typical for sialolithiasis, which were mistaken for salivary calculi based on image findings, are presented. Conclusions: This report intends to highlight the pitfalls in the imaging of parotid gland diseases. Even if malignant tumors of the parotid gland with calcifications are extremely rare, in ambiguous cases, differential diagnoses should be considered carefully. A high suspicion index of the need for further diagnostics in cases with calcifications is practical and could include missing periprandial symptoms, no obstruction signs in the proximal duct, and missing evidence of sialolithiasis in sialendoscopy. Although uncommon, sialolithiasis is the most frequent cause of obstructive and chronic inflammatory symptoms of the salivary glands, with an incidence of 1:10,000 to 1:30,000 inhabitants . The diaIn the following, we would like to highlight the pitfalls in the differential diagnosis of sialolithiasis of the parotid gland, presenting two rare cases of calcified malignant tumors (adenoid cystic carcinoma (AdCC) and acinic cell carcinoma (ACC)), which were mistaken for salivary stones.A 37-year-old male patient presented to our department with permanent swelling and recurrent painful episodes in the left parotid gland without periprandial association or other symptoms. Apart from a testicular tumor in the past, the patient did not suffer from any other diseases. The ultrasound examination revealed a hyperechoic reflex with dorsal sound shadowing in the region of the distal part of the parotid duct matching the diagnosis of sialolithiasis a. No sigA 47-year-old female patient presented to our hospital with recurrent episodes of painful swelling in the left parotid region without periprandial association. The patient had already been treated with multiple antibiotic therapies due to suspected sialolithiasis in another hospital without any relief of symptoms. Her medical history was unremarkable except for chronic nicotine abuse. Our first ultrasound examination initially suggested sialolithiasis with intraparenchymal location a. TherefGland-preserving, minimally invasive interventional treatment options, such as sialendoscopy, intra- or extraductal lithotripsy, or a combined procedure, became the gold standard at large salivary gland centers in the treatment of salivary calculi of the parotid in recent years . As glanSialolithiasis represents the most common cause of calcification within the parotid gland, while other calcifying lesions of the parotid gland are rare and mostly benign. Differential diagnoses include vascular malformations, calcified lymph nodes ,11, and However, even in the case of excluding a tumor from imaging, a surgical exploration rather than a “blind” ESWL of the finding should be preferred. Conversely, surgical exploration of an inflamed, scarred region with a hidden malignancy bears the risk of opening the tumor, which in turn can lead to a worse prognosis by distributing tumor cells in the surgical wound. Additionally, a two-timed approach contains the difficulty of finding exactly those spots where the tumor was incompletely resected during the first session and increased the risk of facial nerve palsy due to the scarred tissue. Furthermore, the probability of a malignant tumor of the parotid gland is extremely low, as, e.g., AdCC represents only 2% and ACC about 1% of all neoplasms of the parotid gland ,13, whicThese cases show that the most obvious diagnosis is not always the right one. However, intraparenchymal sialolithiasis is definitely more common than calcified carcinomas of the parotid gland, and imaging can reveal no definable tumor mass. Therefore, the therapeutic approach should be considered carefully in each individual case, and rare differential diagnoses, such as malignancies, should also be considered. A high suspicion index of the need for further diagnostics in cases with calcifications is practical and could include missing periprandial symptoms, no obstruction signs in the proximal duct, and missing evidence of sialolithiasis in sialendoscopy. Moreover, in cases where a calcified benign tumor is suspected in imaging, surgical resection should be attempted if there is a low risk for facial nerve injury. Otherwise, a close follow-up for the exclusion of another entity is advisable.


Extra cranial primary meningioma of sinonasal tract is a rare entity. It is often misdiagnosed as nasal polyp.Here we report a case of a primary ethmoid sinus meningioma with extension into the nasal cavity in a 41-year-old man. The tumor was completely excised via endoscopic endonasalapproach and the histologic diagnosis of meningioma was established. The patient was regularly follow up for 12 months without recurrence of the tumor.The final diagnosis of primary sinonasal meningioma is based on histopathology and immunohistochemistry analyses results. The importance of complete surgical resection is undoubted and also is a goodindicator prognosis.The otolaryngologists should be aware of the diagnosis of primary meningioma; despite of its rarity it is considered as a possible cause of nasal obstruction. •Primary extra cranial meningioma is rare and its Histogenesis is still unknown.•Clinical presentation of sinonasal ectopic meningiomas is not specific.•Diagnosis is based on histopathological examination and immunohistochemistry.•Complete resection is not always possible due to complex anatomy of nasal cavity and paranasal sinuses. He was not currently taking any medications and had no specific medical history.He was cigarette smoker. There were no history of fever, rhinorrhea or loss of smell. Past and familial history are not significant.General and neurologic examinations were unremarkable. Local examination showed no local rupture or uplift of the nasal skin. Anterior rhinoscopy showed awellcircumscribedsoft tissue mass filling the right nasal cavity and pushing the septum to the left A-B.Fig. The examination of oral cavity and oropharynx was found to be normal. He had no palpable cervical lymph nodes. In fact, the clinical presentation was not specific.Routine tests were normal.The CT-scan of the nasal cavity and paranasal sinuses showed homogeneously enhancing mass in the right nasal cavity. It was a 27 ∗ 14 ∗ 24 - mm ovoid mass with extension into the right ethmoid sinus superiorlyandwith erosion of adjacent bone .Fig. 2CTMagnetic imaging resonance (MIR) was performed ruling out intracranial connection or intraorbital extension A-B.Fig. The decision was to proceed by an endoscopic approach. No special preoperative preparation was required. The patient was taken to the operating room. He was operated on by a senior ENT surgeon after giving his informed consent. Peroperative findings revealed a friable and vascular mass occupying the right nasal cavity. The mass was completely resected using endoscopic endonasal surgical approach under general anesthesia . The posHistologic examination revealed a well circumscribed growth composed of spindly cells with oval bland nuclei and moderate amount of eosinophilic cytoplasm. No nuclear atypia, mitoses, necrosis or invasion of the soft tissue were seen. Tumor cell vimentin, S100 protein (à verifier) and Ki-67 were positively expressed. Negative staining was seen with cytokeratins, CD34, CD31, CD68 and alpha-1 anti-trypsin. Features were suggestive of a WHO grade I nasal meningioma. The post-operative course was simple with no complications or adverse outcomes.The patient was regularly followed up in our outpatient clinic every three months for a total of three years following the operation. Imaging studies and clinical examinations have not revealed any tumor recurrence.3Meningioma is a nonglialtumor of the central nervous system, representing 24–30 % of all intracranial tumors. It arises from arachnoid ‘cap cells’ or meningocytes that is derived from the neural crest Primary extra cranial meningioma is rare, accounting for less than 1 % of cases The histogenesis is still unknown. They may originate frommeningothelial cells that are displaced into this region during embryogenesis The clinical manifestations of sinonasal ectopic meningiomas reported in literature are nonspecific and usually include nasal obstruction, anosmia, facial pain, nasal discharge, epistaxis and facial deformities Nasal endoscopy usually shows a firm, pink to gray mass filling the nasal cavity. Meningiomas are often well circumscribed, globular or lobulated tumors without infiltration into surrounding tissues CT-scan or MIR is unable to identify the nature of the nasal cavity tumor, but it is essential in determining the location, size and it gives information of the extent and invasion of the tumor; compression and deformation can be observed in adjacent bones. Calcification is rare, but no specificity is present Radiological findings in our case were also nonspecific. Therefore, primary sinonasalmeningiomas have been often misdiagnosed as inverted papilloma or nasal polyps WHO  has classified meningioma into 3 grades; the typical or benign type (Grade I), the atypical with frequent mitosis (Grade II) and the anaplastic type with invasion (Grade III) In our case, the slow growth of the tumor as well as lack of perineural and adjacent structures invasion were in favor of the benign nature.Diagnosis is based on histopathological examination and immunohistochemistry; meningiomas are so immunoreactive to vimentin, EMA  and pancytokeratin. Some meningiomas also show positive reactions to S100 protein, CK7, CK20, Ki-67, synaptophysin, CD34, estrogen and progesterone receptor Surgical resection is the treatment of choice for the sinonasal meningioma, but complete resection is not always possible due to complex anatomy of nasal cavity and paranasal sinuses Endoscopic approach allows satisfactory view of the operative field and complete tumor resection. It is correlated with better prognosis. It is less invasive to the surrounding healthy tissue than classical approach Efficacy of adjuvant radiotherapy is not established, it should be considered in case of incomplete tumor resection or malignant transformation In our case we achieved a complete resection of the tumor and radiotherapy was not needed.Y. Liu, H et al. studied the relationships between histological grade and recurrence in 170 cases; recurrence was found to be 8 % in benign tumor K. Sharma et al. achieved a 5-year disease- free survival rate at 72 % 4Primary sinonasalmeningiomas are rare condition. Most of them are benign; only few malignant meningiomas have been reported.It should be considered as one of differential diagnoses of nasal polyps and inverted papilloma.The diagnosis requires histopathological and immunohistochemical examination. Endoscopic approach is very safe and effective approach to resect these tumors completely..We have no sources of funding for our research.This study is exempt from ethnical approval in our institution.We have obtained a written and signed consent to publish this case reports from the patient prior to submission.MEherzi Abir: conception and design of the study.Omri Malika, Hwass Jihen: acquisition of data.Meherzi Abir, Lahmar Rihab: article drafting.Wassim Kermani, Mouna Bellakhdher, Mohamed Abdelkefi: final approval of the version to be submitted.Not applicable.Dr. Meherzi Abir.We declare that we have no conflicts of interest related to this article.


Nasal mucosal melanoma (NMM) is a rare tumor with a poor prognosis. Although an endoscopic resection of malignant nasal tumor now becomes a treatment of choice when the surgical margin can be secured, it is still controversial. We report three cases of NMM that was successfully removed en bloc with clear margins by endoscopic surgery with temporal transseptal access. Cases of a 78-year-old woman, an 83-year-old man, and an 81-year-old man with NMM arising from the inferior turbinate who underwent endoscopic resection of the tumor are discussed in detail. We made temporal transseptal access using septal mucosal flaps. We moved the nasal cavity-occupying tumor to the contralateral side to create a working space to perform endoscopic en bloc resections. This technique is simple yet effective and less invasive than open craniofacial resections for removing malignancies arising from the inferior turbinate. Nasal mucosal melanoma (NMM) is a rare and highly malignant tumor with a poor prognosis . SurgicaOver the past two decades, the indication for endoscopic surgery for malignant tumors in the nasal cavity has expanded. Some studies have reported that endoscopic resection is comparable to open approaches in correctly selected patients , 7. For Case 1The patient was a 78-year-old female who had been experiencing left nasal bleeding for seven months. An endoscopic examination showed an easy-bleeding tumor occupying the lower part of the left nasal cavity. CT and MRI revealed that the tumor arose from the left anterior inferior turbinate without erosion or bone destruction Figure . An inciEndoscopic resection was performed under general anesthesia Figure -1G. Per Case 2The patient was an 83-year-old male who had been experiencing right nasal bleeding for three months. Nasal examination showed a large tumor protruding from the anterior nasal cavity. CT scan and MRI revealed a tumor in the right inferior turbinate without erosion or bone destruction and septal deviation to the left Figure -2B. An iCase 3The patient was an 81-year-old male who had been experiencing left nasal obstruction for two months. An endoscopic examination showed an irregular tumor occupying the common nasal meatus of the left nasal cavity Figure . MRI revIn this report, we described three cases of rare NMMs that were successfully removed using en bloc resection with negative margins using temporal transseptal access. This technique is simple and less invasive compared with open approaches; therefore, we recommend it as a feasible treatment modality for resectable NMM.Owing to the rarity of NMM, it is difficult to recruit a large enough cohort to compare endoscopic resection with the established open approaches . ConsideIn the present cases, we performed an en bloc resection of NMM of the inferior turbinate using temporal transseptal access, based on previous reports , 9 with We raised a septal mucosal flap, with or without an inferior horizontal incision, on the intact side, similar to that in the TACMI technique Figure . HoweverIn conclusion, en bloc resection of NMM using temporal transseptal access and endoscopic surgery is a feasible method for treating resectable NMM localized in the inferior turbinate. Although there are new options, such as proton beam therapy and targeted therapy for treating NMM, elderly patients may not be eligible for these treatments due to their social and physical limitations. Nevertheless, our method is easy but effective and is worth considering to avoid open craniofacial resections.


Appendiceal mucinous neoplasms are usually seen by accident during investigations or surgeries for other reasons in the abdomen. Low-grade appendiceal mucinous tumors have no specific symptoms.We report an unusual case of a 72-year-old man with low-grade appendiceal mucinous neoplasm, which was found accidently during the investigation of the bladder tumor on a CT scan. Pathological examination of the specimen showed a cystic dilation at the distal tip of the appendix, filled with gelatinous mucus. Microscopically, we found mucinous epithelial proliferation with extracellular mucin.Low-grade appendiceal mucinous neoplasm is an uncommon tumor of the appendix. The incidence of low-grade appendiceal mucinous neoplasm is reported to be 1% of all tumors of the gastrointestinal tract and is found in less than 0.3% of appendectomy specimens. Appendiceal mucinous tumors are usually found with no specific symptoms.Low-grade appendiceal mucinous neoplasms are rare tumors of the gastrointestinal tract. The appropriate management for this tumor is appendectomy. The most feared complication of these tumors is seeding of mucin into the peritoneal cavity, leading to pseudomyxoma peritonei. •Low-grade appendiceal mucinous neoplasm (LAMN) is a rare tumor found in the appendix vermiform.•LAMNs may progress to pseudomyxoma peritonei, which is the most troubling complication.•We report a case of a 72-year-old man with LAMN, found during the investigation of the bladder tumor on a CT scan. This tumor is a rare entity found in 0.3% of appendiceal specimens ,2. The aThis case report has been reported in line with the SCARE criteria 2020 .2A 72-year-old man was referred to Tishreen University Hospital in 2021 to perform a radical cystectomy due to Urothelial carcinoma with muscularis propria invasion, which was confirmed by histopathological biopsy. Laboratory tests showed a low hemoglobin level; 10.2 g/dL with hematuria. Other Routine blood values were normal. The patient medical history shows that he underwent transurethral resection of bladder tumor (TURBT) followed by intravesical BCG  therapy administered for 3 years. The patient has smoked 5 cigarettes daily for the past 40 years. He had never used alcohol. He had no allergy to any drugs. There was no tumor history in his family; particularly no history of bladder tumors. Computerized tomography (CT) of the abdomen and pelvis with contrast was performed. Coronal contrast-enhanced CT image shows a mass occupying almost the entire bladder cavity, in addition to the presence of a heterogeneous mass in the right lower quadrant in the expected location of the appendix with tiny peripheral calcifications A. That m3Low-grade appendiceal mucinous neoplasm (LAMN) is an uncommon tumor of the appendix due to much secretion of mucus and accumulation in the lumen which leads to the cystically vast appendix. The incidence of low-grade appendiceal mucinous neoplasm is reported to be 1% of all tumors of the gastrointestinal tract and is found in less than 0.3% of appendectomy specimens . Appendi4Low-grade appendiceal mucinous neoplasms (LAMNs) are rare tumors of the gastrointestinal tract. The treatment of choice for LAMN is surgical excision. Although LAMN is slow-growing, it has the risk of rupturing either spontaneously or due to surgery, leading to seeding of mucin into the peritoneum, forming pseudomyxoma peritonei, which is the most troubling complication.Not commissioned, externally peer-reviewed.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.No ethical approval was needed for this case report.Not applicable.Rabab Salloum.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.Moatasem Hussein Al-janabi: participated in study design, data collections, data analysis, and writing. Tala Zidan: participated in study design, data analysis, and writing. Eman Zidan: participated in study design, data analysis, and writing. Muhammad Sinan Muhammed: performed this surgery and data collection. Rabab Salloum: in reviewing the manuscript.The authors have no conflicts of interest to declare.


The results of adrenal venous sampling suggested a left-side adrenal origin of hypercortisolism, possibly secreted by the mass in the renal hilum. Histopathology after surgical resection of the mass confirmed an ectopic adrenocortical adenoma, which was responsible for the patient’s Cushing’s syndrome. During the 8-month follow-up after surgery, no recurrence of Cushing’s syndrome was found.We report a rare case of Cushing’s syndrome induced by an ectopic adrenocortical adenoma. A 57-year-old woman was diagnosed with adrenocorticotropic hormone (ACTH)-independent Cushing’s syndrome based on clinical manifestation and laboratory information. She was found to have a mass in the left renal hilum via contrast-enhanced computed tomography (CT). The mass was negative, as seen in somatostatin receptor imaging with  Physical examination revealed centripetal obesity, moon face, buffalo hump, and supraclavicular fat pads. She denied any exogenous corticosteroid use.A 57-year-old woman presenting with weight gain, hypertension, and diabetes for 7 years was admitted to the Peking Union Medical College Hospital. Her vital signs at admission were as follows: blood pressure 176/106 mmHg, pulse 102 beats per minute, weight 84 kg, height 151 cm, and body mass index 36.84 kg/mLaboratory investigations are summarized in 2 in the left renal hilum was detected (99mTc-hydrazinonicotinyl-Tyr3-octreotide (HYNIC-TOC) and 18F-fluorodeoxyglucose (FDG) positron emission tomography (PET)/CT to further differentiate the neuroendocrine tumor and its malignancy. 99mTc-HYNIC-TOC single-photon emission computed tomography (SPECT)/CT showed the mass was negative with somatostatin receptor imaging (18F-FDG PET/CT (max) of 2.2, indicating a benign tumor.Abdominal contrast-enhanced computed tomography (CT) demonstrated atrophy of bilateral adrenal glands without any nodules or hyperplasia , are important for detecting the ectopic adrenal tumors. However, it is difficult to differentiate an ectopic adrenocortical adenoma from urothelial or renal neoplasms preoperatively, especially in patients lacking hormonal abnormalities . MisdiagThe treatment of functioning ectopic adrenal tissue is radical resection, and nonfunctioning ones can be managed with the watch-and-wait approach . However131I-6-β-iodomethyl-norcholesterol and 75Se-selenomethyl-norcholesterol, might be helpful to diagnose the ectopic functional adrenocortical tumor, although it is not widely used [68Ga-pentixafor, a CXCR4-specific PET tracer, was reported to exhibit great potential for identifying functional adrenocortical adenomas [Functional imaging could be helpful to obviate the invasive venous sampling for the localization of the responsible tumor in ACTH-independent Cushing’s syndrome. Adrenal scintigraphy with radiolabeled cholesterol analogs, such as ely used ,18. CXCRely used . Recentladenomas ,21. We tadenomas . Howeveradenomas .68Ga-pentixafor PET/CT, may be helpful in diagnosis and confirming the origin of the tumor.In summary, this case report provides a rare case of ectopic adrenocortical adenoma in the renal hilum for clinicians. Selective venous sampling, adrenocortical scintigraphy, and potentially,


We describe a 28‐year‐old man with acute appendicitis associated with gastrointestinal malrotation. The diagnosis was confirmed by a computed tomography scan, and he was treated by laparoscopic appendectomy without a Ladd procedure. Appendicitis in patients with gastrointestinal malrotation is a relatively rare condition. Imaging modalities, especially computed tomography scan, can assist in the accurate diagnosis of this entity. Treatment consists of appendectomy without or with a Ladd procedure. Gastrointestinal malrotations, while often associated with other birth anomalies, are an isolated feature in the majority of adult cases. The estimated incidence in live births of reported intestinal malrotation ranges from 0.03 to 0.5%. The true incidence in adults has not yet been precisely determined. Appendicitis in intestinal malrotation patients is a relatively rare surgical disease.A 28‐year‐old man presented to the emergency department with a persistent dull abdominal pain for about 5 h. The patient stated that pain started in the epigastrium and then spread to the right iliac fossa. Additional symptoms included anorexia and nausea. The clinician suspected acute appendicitis and arranged for routine blood tests and an abdominal ultrasound study. He had an elevated white cell count 18 000/mL), but the ultrasound study failed to identify the appendix. Computed tomography (CT) scan performed subsequently confirmed the diagnosis of acute appendicitis with congenital gastrointestinal malrotation (Fig. 8 000/mL,Gastrointestinal malrotation is a congenital anomaly defined as defective rotation of the middle intestine around the axis of the superior mesenteric artery. However, a variety of anatomical variants have been described.Informed consent was obtained from the patient.


Focal nodular hyperplasia (FNH) of the liver is a benign lesion characterized by hypertrophic nodules with central star-shaped fibrous scars. The etiology and pathogenesis of FNH are not completely understood. A 43-year-old man was hospitalized because of acute abdominal pain. Emergency computed tomography(CT) showed hepatic tumor rupture and bleeding. The patient’s condition improved following arteriographic embolization to stop bleeding. Laparotomy confirmed spontaneous rupture and hemorrhage of focal hyperplasia and the patient remains asymptomatic after an uneventful recovery. FNH with spontaneous rupture and bleeding is extremely rare. Currently, there is no unified management standard for FNH and most previous studies recommend observation and follow-up. We recommend consideration of surgical treatment of cases with spontaneous rupture and bleeding. Focal nodular hyperplasia (FNH) is the second most common benign liver tumor and accounts for approximately 8% of all primary hepatic tumors. It is thought to be a hyperplastic response to increased blood flow in an arterial malformation rather than a true neoplasm , 2. SponA 43-year-old Chinese man was admitted to the hospital because of acute abdominal pain. He had no history of trauma, was in good health, and had been an intermittent alcohol user for about 20 years. Physical examination revealed no palpable mass or tenderness in the right upper abdomen. Blood routine showed that Hemoglobin was 142g/L.Alanine aminotransferase(502.6/U/L)and aspartate aminotransferase(527.9/U/L)were significantly increased. Blood coagulation tests and tumor-marker levels were normal. Abdominal enhanced computed tomography (CT) showed a round, approximately8.0cm×8.0cm mass with a slightly blurred border in the right lobe of the liver, and blood around the liver. A tumor hemorrhage was suspected Figure 1FNH is the second most prevalent benign liver tumor after hepatic cavernous hemangioma. The incidence is highest in those 20–50 years of age, but FNH can occur at any age , 5. It mPrevious cases of FNH with intraperitoneal hemorrhage, including this patient, reported in English-language publications are shown in The occurrence of spontaneous rupture and bleeding of FNH might be explained as follows: Bleeding in FNH patients may be the result of vascular malformations and intratumoral pressure associated , 9, 10. Nguye et al.  describeAt present, there is no consensus on the standard treatment of FNH, which is a benign lesion with no underlying malignancy. Most recommendations are for follow-up of asymptomatic patients . SurgerySpontaneous rupture and bleeding of FNH is very rare, but should be fully considered in patients who experience sudden abdominal pain during follow-up.The original contributions presented in the study are included in the article/Supplementary Material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the participant for the publication of this case report.YS wrote all drafts. BS discussed the meaning of the draft. TZ and KX collect all the references. D-XZ carried out the pathology and collected the clinical data. Y-MH offered conception and finalized the draft. All authors read and approved the final manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Lymphangiomas, or lymphatic malformations (LM), are benign malformations of the lymphatic system characterized by abnormal proliferation of lymphatic vessels. It was first described by Virchow in 1854. They occur rarely in the oral cavity and involve the tongue dorsum more often. Though complete surgical excision is the gold standard and most desirable management, certain limitations restrict this approach. Laser therapy, cryotherapy, electrocautery, sclerotherapy, and intralesional injections of steroids and Bevacizumab are other treatment options in such cases. Here, we present three cases of diverse forms of lymphatic malformations treated with two different modalities of treatment. Vascular anomalies or lesions are congenital, localized structural defects of the vascular system involving arteries, veins, or lymphatics . The mosLymphangiomas, or LM, are benign malformations of the lymphatic system characterized by abnormal proliferation of lymphatic vessels . BlockagVarious classifications have been proposed for lymphangioma: (a) Landing and Farber classified LM into lymphangioma simplex, cavernous lymphangioma, and cystic lymphangioma/hygroma ; (b) basSurgical treatment remains the gold standard. However, there are certain limitations, including chances of bleeding, high chances of recurrences, scarring, especially with large-sized lesions, and esthetic and functional impairment. Furthermore, surgical treatment is limited or non-existent in patients with systemic diseases . ConsideCase 1A female patient aged 14 years reported to our department with a chief complaint of enlargement of the lower lip for four years. The patient gave a history of trauma in the same region for four years, after which the patient developed progressive enlargement of the lower lip. There was no relevant history of any pain, bleeding, or discharge. On inspection, the presence of diffuse enlargement of the lower lip with multiple sessile polypoid growths in the lower labial mucosa was evidenced, extending mediolaterally 1 cm medial from the right commissure of the lip to the left commissure. The mucosa over the growth had small pinpoint spiky projections Figure . The mucCase 2A female patient aged 12 years reported to our department with a chief complaint of enlargement of the dorsal surface of the tongue for three years. There was no history of any pain, bleeding, trauma, or discharge. The patient denied taking any medications. On intra-oral soft tissue examination, evidence of multiple polypoid growths on the left dorsal surface of the tongue was seen. Soft tissue examination revealed the presence of multiple pink to blue papules resembling a pebbly, vesicle-like surface - the so-called "frog-egg" or "tapioca pudding appearance." The mucosa over the growth had small, pinpointed, spiky projections. On palpation, no pulsation was felt. The mass was non-tender and firm, with no pus discharge Figure . The diaCase 3A male patient aged six years reported to our department with a chief complaint of swelling of the left face since childhood. The swelling was gradual in onset and gradually progressing in size. There was no relevant history of any tooth pain, bleeding, or discharge, and medication was appreciated. On extraoral examination, the presence of a dome-shaped swelling was seen, measuring about 6 cm × 6 cm in size. The skin over the swelling was stretched, shiny, and a little erythematous Figure . On palpVarious theories have been suggested for the pathogenesis of lymphangiomas. As per the first theory, congenital LM can be the result of the sequestration of primary lymphatic channels and hence the inability to join main lymphatic vessels or veins. The second theory suggests that the lymphatic channels are unable to drain into the veins, thereby leading to lymphectasia and lymph stasis . The latClinically, superficial lymphangiomas or lymphangioma circumscriptum, appear as papillary/pebbly surfaces with clusters of translucent vesicles with the appearance of "frog-egg" or "tapioca pudding." It may be the same color as the surrounding mucosa or slightly reddish due to secondary hemorrhage. Cavernous lymphangiomas, on the other hand, appear as subcutaneous nodules, masses, or irregular nodularity of the surface with the normal mucosal color of grey or pink projection. Lastly, cystic hygroma, the most common type of lymphangioma, presents as a large, soft, circumscribed swelling with cyst-like cavities containing watery fluid. Cystic hygroma typically occurs on the neck, axilla, or groin. Intraorally, lymphangiomas affect the tongue (most commonly), labial mucosa, buccal mucosa, palate, and gingiva (very rarely) .The diagnosis of lymphatic malformation/oral lymphangioma is clinical. Imaging modalities like color Doppler can help in identifying the condition. Ultrasonography can be used for the detection of the cystic nature and fluid component of the lesion, while angiography can be used to rule out vascular lesions. Biopsy remains the confirmative diagnosis .Surgery is the preferred modality for the complete removal of lymphatic malformations. Surgical excision is mostly associated with recurrence if not removed completely. The recurrence rate varies from 10% to 53% . MoreoveSclerotherapy involves the injection of a sclerosant that causes tissue irritation, endothelial damage, inflammation, and local tissue necrosis. Fibrosis and contracture eventually cause the lesion to disappear . BecauseSodium tetradecyl sulfate or sodium l-isobutyl-4-ethyl octyl sulfate is a synthetically manufactured long-chain fatty acid, commonly used as a synthetic surfactant (soap), whereas bleomycin is an anti-tumor agent, which is a mixture of cytotoxic glycopeptide antibiotics isolated from a strain of Streptomyces verticillus. Bleomycin, through its non-specific inflammatory process, causes fibrosis and the resolution of the lesion ,15,16.Similar to our case, many studies have supported the use of sodium tetradecyl sulfate and bleomycin in the management of oral lymphangiomas ,16. TiwaIt is important to recognize these lesions early to achieve the best results and prevent recurrences. A multidisciplinary approach should be taken in the management of lymphatic malformations owing to their diverse presentations. Macrocystic lesions and lesions that are extensive warrant sclerotherapy as a better treatment modality compared to surgery. Finally, patients with any type of lymphatic malformation should be monitored on a regular basis because recurrence has been reported.


Gastrointestinal (GI) angioectasias/angiodysplasias are the most frequent vascular lesions of GI tract, responsible for ∼5 to 6% of GI bleedings. It commonly involves the small bowel, making it difficult to diagnose and manage endoscopically. Though medical management has been used to prevent bleeding, it has only a limited role in acute severe hemorrhage. In such cases, surgical resection remains the only practical option. However, multiple lesions pose a unique challenge, as resection may not be advisable for long length of bowel involvement. Here, we report a case of recurrent GI bleeding due to multifocal small bowel angioectasias who was managed by a novel technique of full-thickness transmural sutures under intraoperative enteroscopic guidance. At 6 months follow-up, no new bleeding episodes were observed. Gastrointestinal angioectasias (GIAs)/angiodysplasias are the most frequent vascular lesions of GI tract and are responsible for ∼5 to 6% of GI bleedings.2), who was pale looking and tachycardic (heart rate—110/minute) with multiple painless soft tissue swellings all over his body . He had no pain abdomen, vomiting, fever or any history of trauma. Additionally, the patient had also noticed multiple painless subcutaneous swellings all over his body (larger ones being over the neck and chest), since childhood. Examination revealed moderately built and nourished male , somatostatin analogue , or thalidomide.Bleeding of small bowel angioectasias poses a major challenge in view of location and multiplicity, and is difficult to manage with endoscopic intervention. Although resection appears to be a definite management option in cases where surgery is required, in presence of diffusely distributed lesions throughout the small bowel, it may not be an advisable approach. Here, we report such a case of multifocal small bowel angioectasias, who was managed surgically with multiple full-thickness sutures under intraoperative enteroscopic guidance, with good outcome. This case highlights the effectiveness of this innovative surgical procedure without any added morbidity of extensive bowel resection.


PTPN11 mutation. Serial imaging revealed slow tumor regrowth over a three year period which prompted a second resection. The recurrent tumor displayed a similar low grade-appearing histology and harbored the same H3 K27M and PTPN11 mutations as the primary. While the prognostic importance of isolated H3 K27M in spinal gliomas is well-known, the combination of these two mutations in spinal low grade glioma has not been previously reported. Importantly, PTPN11 is a component of the MAPK signaling pathway. Thus, as building evidence shows that low grade-appearing gliomas harboring H3 K27M mutations along with BRAF or FGFR1 mutations have a relatively more favorable course compared to isolated H3 K27M-mutant midline gliomas, the present case provides new evidence for the prognostic importance of activating mutations in other components of the MAPK signaling pathway. This case further highlights the importance of clinico-radio-pathologic correlation when incorporating evolving genetic data into the integrated diagnosis of rare neuroepithelial tumors.We present the case of a 41-year-old man who developed worsening mid-thoracic back pain and imaging revealed a well-circumscribed intramedullary tumor in the thoracic spinal cord. Subtotal resection was performed, and histopathological analysis showed a cytologically bland, minimally proliferative glial neoplasm. Sequencing revealed H3 K27M and an activating The online version contains supplementary material available at 10.1186/s40478-022-01340-9. HIST1H3A and H3F3A genes, respectively. The epigenetic reprogramming as a result of these histone mutations are thought to promote oncogenesis [Mutations in histone genes resulting in H3 K27M are recurrently found in diffuse midline gliomas (DMGs), which are aggressive, high grade glial neoplasms that most commonly occur in children. The defining mutations result in an amino acid change of lysine to methionine at the 28th amino acid (p.K28M) of H3.1 or H3.3 proteins, encoded by PTPN11 is a proto-oncogene tightly linked to regulation of the RAS/MAP-Kinase pathway. Germline mutations in this gene are seen in Noonan Syndrome, an autosomal dominant multiple congenital anomaly syndrome associated with a wide range of malignancies, including neuroblastoma and leukemias [PTPN11 are linked to several solid tumors and hematologic malignancies [PTPN11 mutations have also been detected in a variety of neuroepithelial tumors, including glioblastoma, oligodendroglioma, pediatric low grade gliomas (LGG), and rosette-forming glioneuronal tumors [eukemias . Somaticgnancies . While rl tumors –10.PTPN11 mutations.Here, we present a case of a 41-year-old male presenting with worsening mid-thoracic back pain, and diagnosed with a spinal neoplasm with low grade histopathological features and harboring both H3 K27M and The patient, a 41-year-old man, initially presented with a six-week history of worsening mid-thoracic back pain about 16 months after being involved in a motor vehicle accident as a pedestrian. At the time of the motor vehicle accident, a full skeletal survey demonstrated a non-displaced proximal fibular fracture, nasal bone fractures, bilateral thigh hematomas and nonspecific mildly enlarged lymph nodes. The patient reported no back pain at the time of the accident, and limited spinal imaging demonstrated no acute fracture or subluxation in the cervical spine. The patient left the hospital within one day and recuperated at home with the assistance of outpatient physical therapy.At presentation, the patient described the back pain as constant, dull, and non-radiating, and worsened with prolonged sitting. The patient reported no weakness, numbness, tingling or other focal neurological deficit. While physical therapy along with cyclobenzaprine and meloxicam provided temporary relief, the pain recurred and worsened which prompted imaging. Thoracic MRI revealed a well-circumscribed expansile intramedullary thoracic cord lesion, extending from T8–9 to T10–11 and measuring 5.3 cm in largest diameter, with no clear evidence of cord edema or cystic component . While some histopathological similarities to subependymoma were noted, considering the genetic findings, the case was described as a “Subependymoma-like tumor, with H3 K27M mutation”.Microscopic examination revealed a glial neoplasm with modestly pleomorphic nuclei which were loosely arranged into clusters and embedded within a course fibrillar background Fig. c. A subsOver the next two years, the patient remained clinically stable, and no evidence of tumor growth was seen on initial imaging studies. That said, 28 months after the initial resection, imaging revealed interval growth Fig. : Fig. 1APTPN11 and H3F3A. Whole Genome DNA methylation profiling using clinically validated brain tumor classification [conumee package showed flat copy number profile and unmethylated MGMT promoter. Interestingly, when DNA methylation data were analyzed using T-distributed Stochastic Neighborhood Embedding (tSNE) analysis, the tumor clustered between K27M and MYB low grade glioma clusters  and PTPN11 (p.Q510H) mutations.In this report, we present a case of a low grade glial neoplasm occurring in the thoracic spinal cord which harbored both H3 K27M  Sagittal T1 post-contrast MRI at 32 months demonstrating new contrast enhancement (B) Debulking of recurrent intramedullary tumor with no clear margins from spinal cord. (C) Extensive tumor debulking with improved mass effect on the spinal cord.


Giant cell tumor (GCT) is a benign bone tumor typically seen in epiphysis or metaphysis of mature long bones. Multiple large multinucleated giant cells dispersed among mononuclear spindle cells and monocytes constitute characteristic histological background of GCT of bone (GCTB).A 15-year-old girl was admitted to our hospital with the complaint of pain and swelling in the left leg with difficulty in walking for 2 years. On X-ray of the left leg, osteolytic, expansile, eccentric lesion with sclerotic bone margin on the diaphysis of the tibia was seen suggesting oesteofibrous dysplasia. MRI demonstrated findings compatible with adamantinoma. The subsequent histology report was rather surprising, consistent with giant cell tumor of the bone. Extended intralesional curettage was done with the help of a high-speed burr followed by chemical cauterization and bone grafting. The patient was followed up for 2 years. The patient could walk normally without assistance or any signs of a recurrence.GCTB commonly affects people in their third and fourth decades of life and involves epiphysis of the long bone, but this is a case of diaphyseal GCT, at an age of 15 years. It is challenging to diagnose GCT, if present in an unusual location, unless confirmed by histopathological examinations.A multi-disciplinary approach is required to correctly reach the diagnosis of GCT when it happens to be in an uncommon location(s). Early diagnosis with appropriate treatment and long-term follow-up is mandatory for the successful outcome of the treatment. •GCTB commonly affects people in their third and fourth decades of life and involves epiphysis of the long bone.•We report an unusual case of a 15-year-old girl diagnosed as a giant cell tumor in the diaphysis of the tibia.•Early diagnosis, appropriate treatment, and long-term follow-up are essential for a successful treatment outcome.•Extended intralesional curettage was performed with the help of a high-speed burr followed by chemical cauterization and bone grafting.•After surgery and close monitoring at regular intervals for 2 years, the patient was able to walk normally without any signs of recurrence. They are locally aggressive and potentially malignant tumors. They typically affect young individuals in their third and fourth decades of life, with female predominance However, there is some tumor-like Adamantioma and oesteofibrous dysplasia of bones that are commonly encountered in the diaphysis of the tibia We report a case of a 15-year-old girl who presented to our institution with chronic leg pain and difficulty in walking for 2 years and was later diagnosed to have a giant cell tumor in the diaphysis of the tibia. More interestingly, this case also created a diagnostic dilemma for the clinicians due to its age, location, and radio graphical appearance. As per literature search, diaphyseal giant cell tumor in a skeletally immature patient has only been described thrice in the literature including the last made by el Shamly et al. 2This work has been reported in the line with SCARE 2020 CRITERIA 3A 15-year-old girl came to our hospital when she started experiencing left leg pain while walking to school from her home on a daily basis which was initially gradual in onset, dull aching, intermittent and localized on the anterolateral aspect of the middle part of the left leg for one and a half year. The pain was aggravated by movement and relieved by analgesics. She described the trauma of falling to the ground a year prior, after which her pain increased from mild to severe, getting worse at night. She also noticed swelling in the same region for the last 1 year which is gradually increasing in size. She could move around only with the aid of a crutch. She had no history of fever, anorexia, weight loss, cough, hemoptysis, or any other constitutional symptoms. Her milestones of development are within the normal range. A general examination revealed no abnormality. Her vitals were normal with pulse = 76 bpm, blood pressure = 100/60 mmHg and respiratory rate = 17/min. On loco-regional examination, there was an oval tender swelling on the anterolateral aspect of the middle part of the left leg, measuring 6 × 7 cm, the temperature of which was raised with firm consistency, smooth surface, ill-defined margin, free to overlying skin but fixed to the underlying structure with no regional lymphadenopathy and intact distal neurovascular status. There was no presence of any discharging sinus or any muscle wasting around that area. Gait was antalgic but the range of motion of knee and ankle joints were within normal limit. Systemic examination revealed no abnormality.Her initial laboratory investigations were done. On complete blood count, her Hb = 12.6 g/dL, WBC count13630/cu.mm, C- reactive protein was 2.16 mg/L. Her Parathyroid Hormone level = 120 pg/mL, S. calcium = 9.6 mg/dL, S.TSH = 1.22 μIU/mL, S. Inorganic phosphate = 2.5 mg/dL, S.LDH = 198 U/L, Alkaline Phosphatase = 86 U/L were within normal range and Mantoux test was negative. X-ray of chest posterior-anterior view revealed a normal study.X-ray of the left leg as shown in MRI of the left leg with different planes is shown in A core-needle biopsy of tissue from the left tibia was performed and sent for histopathological examination. The finding was quite surprising to us as it revealed the presence of a large number uniformly distributed multinucleated giant cells interspersed with sheets of round to oval mononuclear stromal cells without the presence of any cystic structure compatible with giant cell tumor of bone as shown in Therefore, we decided to go for the operative procedure. All the pre-anesthetic workup was completed. Under spinal anesthesia, exploration and curettage were done by high-speed burr followed by chemical cauterization with 5 % phenol and 70 % alcohol followed by H₂O₂, shown in Reddish brown fleshy mass was found per operatively as shown in The patient was evaluated after one and half months, 3 months, 6 months, 1 year and 2 year of surgery , giant cell reparative granuloma, giant cell-rich osteosarcoma, and chondroblastoma Using magnetic resonance imaging (MRI), one may determine how far a tumor has infiltrated the soft tissue and bone marrow. The tumor's appearance on T1-weighted imaging is decreased signal intensity, while tumor appearance on T2-weighted images is increased signal intensity, and shows enhancement on gadolinium-enhanced images. While such findings in MRI are shared not only by GCT but also by other bone tumors like adamantioma and osteofibrous dysplasia. This justifies making a diagnosis only after histologically ruling out other possibilities GCTB appears dark brown to reddish in color and friable in nature. The histological appearance of GCTB is characterized by the presence of multiple large multinucleated giant cells dispersed in the background of mononuclear spindle cells and monocytes. Similar to the radiographic variability seen in these lesions, the histological appearance can also be extremely diverse and include areas of cystic degeneration, hemorrhage, hemosiderin deposition, and occasional mitotic figures , or increased spindle cell stroma. Mononuclear spindle cells make up the GCTB's malignant cells. The mononuclear stroma cells are thought to be principally responsible for the formation of the giant cells via the receptor activator nuclear factor K—B ligand (RANKL) Surgery is still the gold standard therapy for this pathology. The surgical options available today range from intralesional curettage with bone cement or bone grafting, cryotherapy to marginal resection, extensive local resection, or en-bloc resection. However, there is a paucity of trials evaluating the different options Besides that, adamantinoma, osteofibrous dysplasia, and osteofibrous dysplasia-like adamantinoma are common related disorders that mostly affect diaphysis of the tibia A benign fibro-osseous disease that causes deformities in children called osteofibrous dysplasia has a significant propensity to affect the midshaft of the tibia, either with or without the involvement of the fibula. In the series of 80 cases reported by Park et al., 77 involved the tibia and three the fibula. In nine of the cases, both the tibia and fibula were involved on the ipsilateral side. Other reported sites of involvement are the ulna and the radius 5Though GCTB commonly involves epiphysis of the long bone, it may sometimes also involve unusual locations like the diaphysis of the long bone. Besides, radiological examination, histopathology from tissue biopsy is the court of appeal for the confirmatory diagnosis and plan the further management accordingly. Early diagnosis, appropriate treatment, and long-term follow-up are essential for a successful treatment outcome. Furthermore, it is imperative to educate patients about the presentation of local invasion or metastasis, so that they can pursue early medical advice. In our case, the patient had no complaints and could walk normally without any signs of recurrence, after surgery and meticulous evaluation at regular intervals for 2 years. However, as planned, the patient will be evaluated every year.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.Nothing to declare.This research did not receive any grant from funding agencies in the public, commercial, and non-profit organization.HM was the chief surgeon and performed the entire surgery with his team.PY conceived, designed and took entire responsibility for publishing this study under supervision of HMPY and SY helped in drafting and editing the manuscript.HM and MK reviewed the manuscript.All authors provided intellectual input to the study and approved the final version of the Manuscript.Prashant Yadav and MD Hasan Masud accept full responsibility for the work and/or the conduct of the study, had access to data, and controlled the decision to publish.Not applicable.Not commissioned, externally peer-reviewed.None to declare.


Contained left ventricular rupture, or pseudoaneurysm, is a rare entity resulting from adhesions confining the defect to a localized portion of the pericardial space. Concomitant infection is even more infrequent. We present the first-known case of a patient with an infected intrapericardial thrombus from a left ventricular rupture. Left ventricular free wall rupture (LVFWR) secondary to acute myocardial infarction (MI) is thought to occur in <1% of cases . Non-traA 64-year-old man without prior medical history experienced an inferior-lateral ST-segment elevation MI. He was treated with drug-eluting stent placement to the right coronary artery that was burdened by a large, acute thrombus. On post-procedure day 2, he complained of angina prompting drug-eluting stent placement to the circumflex artery. Subsequent transthoracic echocardiogram was unremarkable and demonstrated an ejection fraction of 55%. He was discharged on aspirin, atorvastatin, metoprolol and ticagrelor.He returned 5 weeks later complaining of 72 hours of malaise, fevers and chest pressure. He was tachycardic and hypotensive but intermittently responsive to fluid resuscitation. He noted reluctance to present for evaluation, given concerns regarding COVID-19 exposure if he was hospitalized. A computed tomography (CT) of the chest noted a left ventricular irregularity and adjacent thrombus . EchocarEmergent surgical intervention was performed with a median sternotomy, aorto-bicaval cannulation and antegrade cardioplegia. Dense intrapericardial adhesions were lysed, revealing a large pseudoaneurysm sac containing murky, seropurulent fluid. After debridement, a 4-cm defect in the inferolateral wall below the mitral valve was observed . This waPeptostreptococcus asaccharolyticus, supporting a dental source, treated with 6 weeks of intravenous ertapenem. The patient was discharged on post-operative day 14 and continues to do well 2 years from intervention.Blood and surgical fluid/tissue cultures revealed LVFWR occurs in <1% of MI because of early percutaneous revascularization. Patients with LVFWR yield a mortality up to 60% from the resulting cardiac tamponade . In someGiven the profound risk of mortality, early diagnosis and intervention are crucial. In comparison to 50–60% mortality with medical management alone, surgical therapy risks mortality around 10% and lower if without mitral valve intervention .Peptostreptococcus. Staphylococcus and Streptococcus species predominate as the organisms in the referenced reports [Peptostreptococcus outside of recent dental work or clinically observed dental infection represents a rare, but possible source.An infected thrombus is unique as limited available reports describe infection of true ventricular aneurysms . Even le reports . The preA new phenomenon may be observed with the current case related to the COVID-19 pandemic as the patient delayed pursuit of care out of concern for infectious exposure. Up to 42% of USA adults reported postponing or avoiding medical care during the pandemic and 16.9% of adults would avoid emergency care in scenarios of acute MI out of concern for COVID-19 exposure . This fiNone declared.None.