Maximal safe resection is the goal of insular glioma surgery. The combination of intraoperative augmented reality (AR) diffusion tensor imaging (DTI) fiber tracking with fluorescein dye (F) helps achieve this goal throughout a microscope-based visualization of the tumor and white matter fiber tracts.The aim of the present video article was to show the technical key aspects of DTI-F microscope-based AR-assisted surgery during the gross-total resection of an insular Berger-Sanai type I+IV high-grade glioma in a 63-year-old patient, performed through a pterional transsylvian approach.https://stream.cadmore.media/r10.3171/2021.10.FOCVID2157The video can be found here:  This video article aims to show the technical key aspects of the microscope-based augmented reality (AR) with diffusion tensor imaging (DTI) and fluorescein in insular glioma resection.A 63-year-old female patient suffered from a progressive decline in learning and memory, and new-onset epilepsy. The neurological exam revealed a neurocognitive and visuospatial impairment. Past medical history was unremarkable.MRI revealed a right insular Berger-Sanai type I+IV high-grade glioma.The tumor arose at the level of the extreme capsule, and it involved the short and long gyri.The claustrum and the lentiform nucleus were pushed medially, and the medial part of the lesion was in contact with the putamen.1–9In their progressive growth, insular gliomas tend to encase the lenticulostriate arteries, this aspect being the major cause of morbidity of insular glioma surgery.3D-rendered MRI showed in more detail the anatomical relationships between the tumor, circular sulcus of the insula, and putamen.Preoperative DTI fiber tracking showed the spatial distribution of the corticospinal tract, occipitofrontal fasciculus, posterior thalamic peduncle, and optic radiation compared to the tumor. It also allowed for the selection of the surgical approach.A pterional transsylvian approach was selected.Noteworthy, transsylvian route allows for multiple working corridors.The different vascular supply of the basal ganglia and insular cortex from the lenticulostriate arteries and MCA, respectively, is to be taken into account during the planning of surgery.Total intravenous anesthesia target-controlled infusion was used to allow the neuromonitoring. Cortical subcortical mapping was performed with transcranial electric stimulation motor evoked potentials, while subcortical mapping was carried out with the high-frequency bipolar stimulation technique.Asleep craniotomy was performed. A one-layer submuscular pterional approach was carried out with the full exposure of the lateral part of the sylvian fissure.Brain mapping is recommended since DTI provides only anatomical information but no data about the function.The first step of surgery consisted of the opening of the carotid cistern, Liliequist membrane, chiasmatic cistern, along with the fenestration of the lamina terminalis.Meticulous sharp dissection of the arachnoid membranes is of utmost importance in avoiding any damage to the neurovascular structures of the basal cisterns.It is also essential to keep clean the surgical field.A wide opening of the basal cisterns, Liliequist membrane included, allows for CSF withdrawal. This is the key point to avoid the use of fixed brain retraction in the transsylvian approach.The dynamic retraction by the surgical instruments is less traumatizing and more effective in allowing for the progressive recognition and exposure of the anatomical structures.The visualization of the lenticulostriate arteries is crucial since they mark the most medial limit of surgery.Microscope-based AR fiber tractography provided for virtual view of the anatomical relationships between the lateral lenticulostriate arteries and white matter fiber tracts.The splitting of the lateral and anterior insular compartment of the sylvian fissure and opening of the sylvian cistern allowed to expose the M1 segment of the middle cerebral artery (MCA), MCA bifurcation, superior and inferior limiting sulcus of the insula, limen insula, and entire insular cortex.In this case, the middle trunk of the MCA originated from the superior trunk.The insular cortex showed a fluorescein enhancement.The cortical branches of the MCA were coagulated and the tumor was resected in piecemeal fashion. The use of fluorescein filter aided the recognition of the tumor boundaries.Care must be taken during tumor removal in avoiding mechanical vasospasm of the MCA or its branches caused by an excessive retraction of the vessels.The use of the cavitron ultrasonic aspirator facilitated the tumor resection.DTI allowed for a constant awareness of the spatial location of the corticospinal tract, fronto-occipital fasciculus, and optic radiation.The accuracy of the AR fiber tractography was checked at a cortical and subcortical level, having the motor strip as a landmark. At the cortical level, the matching between the virtual corticospinal tract and the opercular part of the motor area was tested. At the subcortical level, the matching was verified through the subcortical mapping.DTI navigation was extremely useful during the resection of the deepest part of the lesion, close to the putamen.At this step, the lack of fluorescence confirmed the gross-total resection of the tumor.At the end of surgery, the check of the blood flow ruled out spasm of the MCA and its branches.ICG videoangiography showed the patency of the MCA.A gross-total resection of the glioma was achieved, as confirmed by the postoperative MRI. Postoperative DTI documented the preserved structural anatomical connectivity of the corticospinal tract, IFOF, and optic radiation. It was basically as an anatomical confirmation of a functional check which has been already obtained intraoperatively by means of the cortical-subcortical mapping of the motor pathway.The patient was discharged without deficits on the 7th postoperative day. The relatively longer perioperative follow-up was justified by the known risk of delayed ischemic complications. A urinary tract infection occurred at the 3rd postoperative day, required the administration of intravenous antibiotic therapy. Pathology revealed an anaplastic astrocytoma. Standard Stupp protocol was employed and no recurrence occurred at the 21st-month follow-up.In conclusion, DTI-fluorescein microscope-based AR resulted safe and effective in maximizing the extent of resection of the reported insular high-grade glioma. Its association with brain mapping is recommended. Further theoretical advantages may also come from its use during awake surgery.We thank Eng. Giorgia Di Giusto for the technical support.The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this publication.Primary surgeon: Luzzi. Assistant surgeon: Giotta Lucifero. Editing and drafting the video and abstract: both authors. Critically revising the work: Luzzi. Reviewed submitted version of the work: Luzzi. Approved the final version of the work on behalf of both authors: Luzzi. Supervision: Luzzi.


Paraganglioma of genitourinary tract is uncommon, and origin from ureter is even rarer. We aim to present a case of paraganglioma from ureter in a 48-year-old female patient, who presented with gross hematuria.We present a 48-year-old female who complained of gross hematuria for one week. A left ureteral tumor was found by image study. However, hypertension was unexpectedly recorded during diagnostic ureteroscopy survey. Due to persisted gross hematuria and bladder tamponade, she underwent left nephroureterectomy with bladder cuff resection. Blood pressure surged again when the tumor was surgically approached. Ureteral paraganglioma was confirmed according to pathological report. After the surgery, the patient recovered well, and no more gross hematuria was noted. She is now under regular follow-up at our outpatient clinic.Ureteral paraganglioma should be kept in mind not only when blood pressure fluctuates during operation, but also before we manipulate the ureteral tumor when gross hematuria is the only sign. Whenever the presumption of paraganglioma is raised, laboratory evaluation and anatomical or even functional imaging should be considered. The concomitant anesthesia consultation before the surgery should not be deferred, either. Paraganglioma of urinary tract is rare, and it is reported to mostly originate from bladder, kidney and prostate . Among tA 48-year-old female, who denied any systemic diseases, presented with severe gross hematuria and voiding difficulty for one week and came to the emergent department of our hospital for help.Tracing back her history, she had visited regional hospital for gross hematuria several weeks ago before she came to us. Urinary bladder tamponade was noted and manual bladder irrigation with blood clots evacuation was also performed. Further imaging with contrast-enhanced computed tomography (CT) scan showed left hydronephrosis, and a round, well-defined retroperitoneum mass was noted adhering to left upper ureter, lying at left para-aortic region and the aortic bifurcation level. It was measured about 4.3 cm, and displaced the ureter medially, and the ovarian vein anteriorly. It also indented on the psoas muscle. The mass showed homogenously vivid after contrast enhanced. Massive blood clots were found within renal pelvis and bladder. Diagnostic semi-rigid ureteroscopy was arranged, and serpentine blood clots were found within left ureter. However, the ureteroscopy could not pass the compression site, where the sharp angulation existed. Unexpectedly, blood pressure surged when they tried to pass the stricture site with ureteroscope. The diagnostic ureteroscopy was incomplete and double J stent was placed for hydronephrosis. She was then referred to our hospital for further management.At our emergency department, fever up to 39.2’c was recorded, but the blood pressure was within normal range (112/71 mmHg). The accompanied symptoms include dysuria, urinary frequency, and urgency. Basic hemogram revealed leukocytosis and elevated CRP level, but there was no deterioration of renal function. Meanwhile, pyuria was also recorded through urine analysis.Reviewing her CT at regional hospital, we could see left hydronephrosis with a left retroperitoneal tumor about 4.3 cm adhering to and compressing left upper ureter with massive blood clots in bladder Fig. . We alsoFever gradually subsided after adequate antibiotics treatment. However, gross hematuria persisted with intermittent Foley catheter obstructed by blood clots. Manual bladder irrigation with blood clots evacuation was performed several times but in vain. Accordingly, her hemoglobin level dropped from 11 to 8 g/dL, so adequate blood transfusion was also given. During the clinical course before further surgical intervention, her blood pressure was kept within normal range.After discussing with the patient, emergent excision of left retroperitoneal mass, which compressed and adhered to upper ureter, and possible nephroureterectomy with bladder cuff excision was arranged.Under the impression of paraganglioma, though laboratory workup failed to be completed, we informed this situation to the anesthesiologist and made sure blood pressure was under cautious monitoring. During the operation, a round tumor was found adhesive onto left upper ureter, and severe adhesion between the tumor and gonadal vein was also identified. Extremely high blood pressure (> 200 mmHg) was detected during tumor manipulation even after multiple antihypertensive agents given by anesthesiologists. After tumor blood supply ligation, blood pressure restored to normal range rapidly. The patient recovered smoothly after the operation, and no hypertension was recorded after the surgery. The hemoglobin level also came back to normal range. Finally, she was discharged 9 days after the operation.For the surgical finding, we found the specimen was a 4 × 3.5 × 3 cm elastic mass, which attached to left ureter, compatible with CT findings Fig. . MicroscSix months after the surgery, the level of 24 h-urine catecholamines were within normal range. The follow-up computed tomography also revealed no recurrence of retroperitoneal tumor.Paraganglioma, which is also described as pheochromocytoma by previous articles of bladder, has been first described in 1953 by Zimmerman , and it The previous reports showed that the ureteral paraganglioma could be measured from 3.5 to 6 cm and could be located from renal hilum to the site of insertion to bladder.The postoperative follow-up time of all six cases was around 1–2 years, and no tumor recurrence was found according to these studies.For paraganglioma arising from genitourinary tract, bladder is the most common site of growth. The malignancy rate of these tumors has been described as 15–20%.Among these cases of GU tract paraganglioma, they could be adequately managed by surgical excision .The majority of initial presentation is hypertension or related symptoms and can be followed by hydronephrosis. Gross hematuria as initial symptom is relatively rare and differential diagnosis remains a challenge in clinical practice. The uncommon presentation in our case could be explained through the view of histopathology, by which the origin of the tumor from muscle layer of ureter might explain this phenomenon.Since hypertension is the most common symptom in previous reports, it is crucial to raise such a presumption whenever fluctuating blood pressure is recorded when evaluating the disease, especially during manipulation of the tumor. We’ve learned a lesson from our case that although normo-tensive status was detected in most times in the clinical course, blood pressure surged during diagnostic ureteroscopy stood for a critical hint, through which ureteral paraganglioma should be considered.From previous studies, we realized that plasma metanephrines are the most accurate parameter for detection of pheochromocytoma, whose sensitivity can reach 99%, and urinary fractionated metanephrines, with 97% sensitivity, are the most accurate parameters for detection of pheochromocytoma, and they perform better than urinary VMA from our case, whose sensitivity is only 77% . The choIt is well known that pathological evaluation for malignant pheochromocytoma or paraganglioma is difficult. The Ki-67 stain, and Pheochromocytoma of the Adrenal gland Scaled Score (PASS) score is what we can facilitate in order to offer more accurate follow-up . Grossly123I]-metaiodobenzylguanidine single photon emission computed tomography (123I-MIBG SPECT), which provides high specificity for paraganglioma identification, and positron emission tomography (PET) with radiopharmaceuticals such as 6-[18F]-fluorodopamine (18F-FDA) and [18F]-fluoro-dihydroxyphenylalanine (18F-FDOPA). They have also been used to locate paraganglioma and can also be seen as a routine functional imaging of paraganglioma. What’s more, comparing to 123I-MIBG SPECT, [18F]-fluoro-2-deoxy-D-glucose (18F-FDG) PET seemed to detect metastases better with high specificity. The uptake of 18F-FDG could indicate hereditary syndrome that underlies the paraganglioma [After the operation, image follow-up at clinic is important for us to evaluate any signs of recurrence or metastases. We can learn from former studies that the survey and follow-up of paraganglioma include anatomical and functional imaging. The former includes CT and magnetic resonance image (MRI), which could reveal the contour and content with high sensitivity. The latter is composed of 3I-metaio18F-fluorodopamine, 18F-dihydroxy-phenalalinine (DOPA), 18F-FDG or 68Ga-DOTATOC/DOTATATE/DOTANOC. The 18F-FDG stands out with a sensitivity of 80–100%, but with less specificity to detect extra-adrenal disease. Ga-DOTATOC and 18F-DOPA have been shown to be more accurate than 123I-MIBG, especially in small lesions and in extra-adrenal diseases [More recently, the European Association of Nuclear Medicine recommended functional imaging for paraganglioma with ligands like diseases , 14.Preoperative diagnosis of paraganglioma may sometimes fall into dilemma if clinical symptoms are occult and laboratory study is limited. In our intraoperative hypertension is the only hint to indicate paraganglioma, and we also failed to gain the result of urinary VMA due to severe hematuria and we also couldn’t check urine metanephrines due to the limitation of facility. Other prompt and clinically feasible survey of paraganglioma may be needed in the future if urgent surgical intervention is inevitable.Clinical presentation of ureteral tumor with gross hematuria raises the suspicion of urothelial carcinoma other than paraganglioma. However, the differential diagnosis of paraganglioma should be kept in mind if blood pressure surged when manipulating around the main tumor during ureteroscopy or even surgical manipulation. Even if gross hematuria is the only sign at first, this diagnosis of paraganglioma shouldn’t be omitted. Besides, anatomical or even functional study should always be done before invasive procedure if it is clinical available. Careful anesthesia consultation should not be omitted before the surgery. After the operation, the follow-up image study, renal function and urine test should also be monitored.


Elastofibroma dorsi is a rare benign tumor of soft tissue, typically under the lower angle of the scapula. Its specific location and distinctive clinical symptoms can provide enough information for diagnosis. Nevertheless, pathological confirmation by biopsy may be needed to rule out other malignancies.Here, we present two cases of 63-year-old and 49-year-old female Asian patients who came to us with the chief complaint of pain and bulging in their shoulders. Both patients had rubbery and mobile masses. Also, shoulder movements were not restricted in the examination; however, the patients expressed pain during movements. Computed tomography scans were compatible with the diagnosis of elastofibroma dorsi. Surgical excision was performed for both cases owing to the symptomatic nature of the masses, and histopathological findings confirmed the diagnosis.Elastofibroma dorsi is a benign pseudotumor presenting with an uncomfortable feeling in the shoulder with movement in older females. In typical symptom-free cases of elastofibroma dorsi, observation is sufficient, while in symptomatic patients or if there is suspicion of malignancy, complete resection with marginal resection is the treatment of choice. Elastofibroma dorsi (EFD) is a rare, slow-growing, nonencapsulated, benign tumor with soft tissue origin . The preThis study describes two patients: one with a case of bilateral elastofibrolipoma and the other with unilateral fibrolipoma, presenting with shoulder bulging and pain, diagnosed with typical histological appearance. Surgical excision was performed owing to the symptomatic nature of the masses in our cases.A 63-year-old female Asian patient was referred to our hospital with a chief complaint of bilateral shoulder bulging and, also, right-sided shoulder pain, especially during shoulder movements. On physical examination, bilateral well-defined masses were detected in the subscapular area, of which the right-sided mass was larger. Both masses were rubbery and mobile. Also, shoulder movements were not restricted in the examination; however, the patient expressed pain during movements. Contrast material-enhanced CT demonstrated a soft-tissue mass. The borders of the mass were indistinct, but adjacent fat planes were preserved. The majority of the mass had an attenuation similar to that of the adjacent skeletal muscle; however, scattered microscopic linear fat attenuation was seen centrally throughout the mass. There were no nodular areas of contrast enhancement, no calcification, and no adjacent osseous destruction. No tissue sampling or further imaging was deemed necessary for diagnosis, since these were classic imaging findings and the location was typical for elastofibrolipoma. Figure Surgical excision was performed owing to the symptomatic nature of the mass at the right side. In lateral position, under general anesthesia, the chest wall skin, fascia, and muscles were opened, and an 8 × 10 cm mass was completely excised. The resection was done until the margin where the rubbery consistency of the mass was palpated. It is possible that some parts of the surrounding fibrous tissue remained. A Jackson–Pratt (JP) drain was inserted, and subsequently, the chest wall was closed. Also, the mass was sent for pathological evaluation.The mass was halved during resection, and two irregular, semi-ovoid, gray-yellowish, rubbery, solid masses were received by the pathologist: one was 5.5 × 4 × 2.5 cm with attached soft tissue of 5.5 × 2 × 1 cm, and the other one was 3 × 3 × 2 cm with attached soft tissue of 3 × 3 × 1 cm. The cut section of the larger mass showed a white-yellowish solid area, and the cut section of the smaller mass showed a homogeneous yellowish area without hemorrhage or necrosis. Microscopic examination showed paucicellular tissue with thick densely eosinophilic elastin bands, which were positive with Verhoeff elastin stain  reports can show more specific findings. In particular, MRI is capable of showing expansile, solid, non-encapsulated, and heterogeneous tumor. Entrapped fat within a predominantly fibrous mass is demonstrated on T1- and T2-weighted sequences by predominant signal in comparison with muscles, which is an indicator of fibrous tissue and is typically intermingled with hypersignal lines representing fat tissue , 20.When the lesion appears as heterogeneous, indistinct, and non-encapsulated infra- or subscapular soft-tissue mass, isoattenuating to muscle (fibrous tissue) with strands of fat attenuation, CT can be diagnostic. However, the lesion can also appear homogeneous when small –23. ThesIn case of suspicion of malignancy or the presence of symptoms, confirmation by biopsy is needed. Excisional biopsy is the preferred method of confirming the diagnosis and excluding malignancy –11. The Since there are no reported malignant transformations, surgery in asymptomatic patients is not indicated, and observation of the patient is enough. In symptomatic patients, surgical excision with marginal resection is sufficient. Radiotherapy can be used if lesions are unresectable owing to their location, if the lesions recur because of incomplete resection, or if patients are at high medical-anesthetic risk .Elastofibroma dorsi is a benign pseudotumor presenting with an uncomfortable feeling in the shoulder with movement in older females. In typical symptom-free cases of elastofibroma dorsi, complementary studies are necessary, and observation is sufficient. In symptomatic patients or if there is suspicion of malignancy, complete resection with marginal resection is the treatment of choice.


Nearly 20% of all mesenchymal tumors are liposarcoma in origin, mostly occurring in extremities and trunk. However, few cases of liposarcoma in the hypopharynx have been documented. This atypical localization of liposarcoma warrants a great interest in reporting such a case. Here, we report an 81-year-old Saudi male who presented to our clinic complaining of progressive dysphagia and globus sensation for two months. On examination, using a flexible nasopharyngoscopy, a hypopharyngeal mass occupying the left piriform sinus originating from the mucosa of the posterior pharyngeal wall and anteriorly from the anterior and medial piriform sinus mucosa was observed. Contrasted head and neck CT-scan revealed a benign-looking well-defined left-sided submucosal cyst aligned along the left aryepiglottic fold encroaching and narrowing the laryngeal inlet with dimensions of 1.8×2.1×2.7 cm. The mass was resected successfully using a trans-oral approach. A histopathological review showed spindle stromal cells that reacted positively for CD34 (Qbend10) on immunohistochemical staining and positive result for MDM2 (12q15) Amp. The pathology result indicates an abnormal amplification of the MDM2 gene region. The patient was followed for almost two years without evidence of recurrence. In conclusion, atypical lipomatous tumors (ALTs) of the hypopharynx are rarely diagnosed, and the gold standard for diagnosis is biopsy. Transoral endoscopic approach has a better outcome than cervical approach. Follow-up of patients with ALT is crucial, due to the highly recurring nature of the disease. Here we present a rare case of ALT, the patient had complete remission without complication. Liposarcoma is a malignant tumor of adipose tissue, most commonly seen in the extremities, trunk and retroperitoneum -2. RarelALT is a well-differentiated type of liposarcoma that has a favorable outcome if treated surgically . LiposarALT is differentiated from GFVP histologically, they present as variable size adipocytes, atypical multivacuolated lipoblast, and nuclear atypia in adipocytes and spindle cells . The impHere we present a case of an 81-year-old male, a former soldier at the Saudi National Guard, who presented to King Abdulaziz Medical City (KAMC) in Riyadh, Kingdom of Saudi Arabia. He was referred to our service complaining of difficulty swallowing solid food for the past two months, with a progressive deteriorating course. He chokes on food residue and complains of a bothersome globus sensation. The patient has a negative history of pneumonia, dysphonia, structural pharyngeal dysphagia, and B-symptoms . Past medical history was significant for hypothyroidism, hypertension, heart failure, anteroseptal MI, dyslipidemia, reflux esophagitis, gastric ulcer, right ear hearing impairment and chronic kidney disease.On physical exam using nasopharyngoscopy, a hypopharyngeal mass was seen occupying the left piriform sinus originating from the mucosa of the posterior pharyngeal wall and anteriorly from the anterior and medial piriform sinus mucosa Figure . The masFurther investigation with a contrasted head and neck CT-scan revealed a benign-looking well-defined left-sided submucosal cyst aligned along the left aryepiglottic fold encroaching and narrowing the laryngeal inlet with no extension beyond the pharyngeal wall. The mass’ appreciated dimensions were 1.8×2.1×2.7 cm  on immunohistochemical staining and positive result for MDM2 (12q15) Amp. This pathological result indicates an abnormal amplification of the MDM2 gene region. The patient was first followed up two weeks post-operatively, was recovering well and symptomatically improving, vocal folds’ mobility was not affected. There was no mass or lesion seen using a 70-degree rigid endoscope  because it is associated with less surgical morbidities and complications , decreased hospital stay and avoidance of tracheostomy. Though surgical complication can occur with transoral endoscopic approach, it occurs in lower frequency than in the trans-cervical approach. Follow-up of patients with ALT is crucial, due to the high recurrence nature of the disease. Here we present a rare case of ALT, in which the patient had complete remission without complication.


Colonic diverticulitis is one of the common causes of surgical intervention in general surgical practice. In most cases, surgical intervention depends on the presence of a collection around the sigmoid colon, the feasibility of percutaneous drainage, and the patient’s medical condition. The collection can occur in the thigh in rare cases due to a fistulous communication with the retroperitoneum and can track gravitationally along the psoas muscle into the thigh and leg without any discernible collection around the sigmoid colon or in the abdominal cavity. We came across a similar case of a 54-year-old man who presented with abdominal pain, and thigh and leg swelling. Left-sided colonic diverticulitis was seen without any discernible abdominal collection and a thigh abscess during the initial clinical presentation. He was treated with multiple drainages of the thigh abscess, ultimately followed by a Hartmann’s procedure over a total hospital admission period of 52 days. Current literature does not throw much light on such a situation and makes it all the more critical to illustrate this case. We present this rare case and give a complete account of investigations, disease course, and the interventions done to throw light on the optimal management of such cases. Colonic diverticulitis is one of the dreaded differentials for left iliac fossa pain and is known to be caused due to low intake of fibers in the diet and an increase in the intraluminal pressure due to motility abnormalities . Left-siHowever, in rare instances, the complications of colonic diverticulitis can be devastating. We present a rare case of sigmoid diverticulitis without any abdominal paracolic collection, presenting with a fulminant abscess in the thigh and masquerading with musculoskeletal complaints rather than abdominal symptomatology. Only a few prior documented case reports in the published literature are available for review ,5,6, and3/mL ) with predominant neutrophilia (22 x 109/L), low estimated glomerular filtration rate (28 ml/min/1.73 m2), and mildly elevated D-dimer levels. His co-morbidities included a high BMI of 45 and bronchial asthma. A 54-year-old male was admitted under the care of an acute medical team with a three-day history of abdominal pain, loose stool, and vomiting. Additionally, he had a two-week history of gradually increasing left thigh swelling originating in the inguinal region and spreading distally. On examination, he had tenderness over his left iliac fossa and a tense left thigh swelling along the posteromedial aspect of his left distal thigh extending up to the medial aspect of the knee joint. Since he was febrile but not in shock, along with erythema and swelling in the thigh and leg, the medical team formulated provisional clinical diagnoses of gastroenteritis along with cellulitis and possible deep vein thrombosis (DVT). Relevant laboratory work-up revealed raised C-reactive protein (CRP) (332.5 mg/L), high white cell counts , hinting toward a bowel-related source of the infection. The patient’s inflammatory parameters started coming down, he was no longer septic, but he did not improve in terms of abdominal pain and continued having intermittent fever episodes. The antibiotic cover over the whole course of treatment included intravenous (IV) co-amoxiclav and clindamycin initially, later followed by a change to IV piperacillin with tazobactam.After a multidisciplinary team meeting (MDT) involving general surgery and orthopaedics, a CT-guided intra-abdominal drainage was requested the same day, along with a decision to drain the thigh abscess on an urgent basis. Radiologists deferred the percutaneous drainage due to the lack of well-formed drainable retroperitoneal collection and further deterioration of the patient’s clinical condition. The thigh abscess drainage was carried out as a priority by the orthopaedic team, along with conservative non-surgical management for his diverticulitis with appropriate antibiotic coverage as decided by the general surgical team. On incising the most dependent part of the thigh abscess at the posteromedial aspect of the distal thigh, there was a gush of foul-smelling pus (2 liters), which was evacuated, and a surgical drain bag was attached to the surgical thigh wound, which was kept open. The patient continued to have abdominal symptoms, and a re-look thigh washout was done two days later. Following the first surgery, there was a continuous drain of up to 100ml pus per day from the incision, which continued even after the second washout. The culture sensitivity on the pus from the thigh wound showed anaerobic infection and isolated bacteria A second CT, which was a contrast-enhanced CT (CECT) to check the progress of the diverticulitis, was done on day eight, which showed an interval regression of the thigh abscess, and a few residual air pockets in the thigh abscess with persistent diverticular inflammatory changes in the left iliac fossa. There was a possible fistulous communicating tract extending from the persistent peri-colonic inflammatory changes to the retroperitoneal left psoas muscle, as seen in Figure 3/mL) in the days after the second surgery, a third contrast-enhanced CT was done on day 18. It showed re-progression of the thigh abscess with the interval appearance of a liquefied thigh abscess, as seen in Figure Since the patient did not improve in terms of abdominal pain, and there was a repeat rise in inflammatory markers , but they were still very high, and the patient’s hospital stay was extended.The orthopaedic team performed a third thigh washout on day 25 due to non-ceasing pus drainage from the thigh incision and interval resurgence of thigh swelling due to re-accumulation of pus within the thigh. Unfortunately, this drainage also did not yield any improvement in terms of abdominal pain. There was a marginal drop in inflammatory markers with CRP (245 mg/L) and white cell counts (18 x 10Repeat MRI imaging on day 36 showed the presence of a few smaller recurrent collections in the medial thigh with infective myositis and again showed persistent inflammatory changes in the left iliac fossa without any demonstrable well-formed intra-abdominal collection, as seen in Figure Due to the patient being no better and the reluctance of the general surgical team to intervene, a fourth thigh washout was done by the orthopaedic team on day 40 with further incisions on the anterolateral aspect of the thigh, coinciding with the collections seen there in the previous MRI with only small pus drainage from these incisions at this setting. The patient started deteriorating and became septic the next day with persistent fever and increasing abdominal pain; hence a fourth contrast-enhanced CT on day 42 for the first time showed an intra-abdominal retroperitoneal collection around the left-sided colon measuring 15 x 6.5 x 3.5cm as seen in Figure This was followed by an urgent Hartmann’s procedure on day 42, following which the patient improved, and no further collections in the thigh were identified. Hartmann’s procedure was technically difficult due to the morbid obesity of the patient, and it revealed an inflamed, thickened segment of the sigmoid colon adherent to the left pelvic brim and left lateral and posterior abdominal wall which required meticulous dissection. There was a perforation identified intraoperatively, but the whole procedure was completed with no untoward incidents. The patient improved remarkably over the next couple of days without any complications and required no further thigh washouts. He was ultimately discharged in a non-febrile state a week later with appropriate antibiotic coverage. The patient’s condition continued to improve over the next couple of weeks, and he was followed up for two months by both the orthopaedic and general surgery team and finally discharged from our care without requiring any further follow-ups. The Hartmann’s procedure done was not reversed by the surgical team on follow-ups due to the risk of complications. Additionally, the posteromedial distal thigh wound was kept open throughout the entire time; it was followed up by the community nurse for a couple of months with regular dressings until it healed with secondary intention.Just before Hartmann’s procedure, during the same anaesthetic, cystoscopy and ureteric stenting on the left side were performed on the table as a prophylactic measure which was removed after the abdominal procedure. The stenting was done to mitigate any problems should the ureter get damaged during the complex surgical dissections required in the procedure. Perforation of sigmoid diverticulitis lands it directly into the complicated category, which is known to have some kind of morbidity associated with it after surgery in 43% of the cases  while haThe case report highlights the problems of delayed abdominal intervention in complicated diverticulitis cases due to the paucity of literature on such cases and hence the lack of awareness about it. Thigh collections originating from other areas such as psoas abscess , post maThe paucity of literature makes managing such cases difficult, with cases often having delayed recovery due to late gastrointestinal surgical intervention in the absence of a well-formed intra-abdominal/retroperitoneal collection. Often such cases are managed conservatively due to the reluctance of the GI surgical team to intervene in the absence of an intraabdominal collection. A good clinical examination followed by a CT scan of the pelvis and abdomen, which is the gold standard investigation, should be followed in each case with such presentations. It has been seen that, along with clinical acumen, multidisciplinary meetings go a long way in improving outcomes -17.In our case, the patient was primarily managed by the orthopaedic department due to the initial presentation of a thigh abscess and the absence of intra-abdominal collection. The immediate thigh abscess drainage as an emergency life-saving measure was warranted in this acutely septic patient; the thigh abscess was possibly recurring due to gravity dependant tracking of the infective fluid along the left-sided retroperitoneum through the communicating fistulous tract into the left proximal thigh and extending into the proximal leg. The patient underwent repeated surgeries to drain recurring thigh abscesses when perhaps post-inflammatory diverticular perforation and secondary fistulisation were possibly the primary source of infection and could have been tackled with an earlier GI surgical intervention. The Hartmann’s procedure eventually done is the gold standard treatment in acute septic diverticular perforation cases .The dilemma now comes about the best timing of abdominal intervention, which in this case, we believe was possible after the second washout of the thigh, when the pus continued to drain. An MDT between the general and orthopaedic surgeons after incomplete response after each washout regarding further surgical intra-abdominal management could have helped in faster recovery. On reflection, the delayed intra-abdominal surgical intervention resulted in a prolonged hospital stay of 52 days and four thigh washouts along with the abdominal surgery, not excluding the patient’s surgical morbidity, increased interval radiation exposure, and additional financial costs to the hospital. Additionally, the patient caught a COVID-19 infection during the prolonged hospital stay but luckily fought through it and recovered uneventfully.Cases with diverticular perforation with uncommon complications need a multidisciplinary holistic approach to improve patients’ recovery and reduce hospital stay and morbidity. A paucity of published literature regarding the atypical and uncommon complications of colonic diverticular perforation itself can complicate matters. As seen, thigh abscesses can masquerade as the initial clinical presentation in left-sided colonic diverticular perforations instead of an intra-abdominal collection, in which case, along with the help of CT, we believe a detailed clinical examination in the first instance followed by continued interdepartmental discussion/communication is the best way forward to decide timely and effective interventions. Additionally, in such non-responding cases after surgical thigh drainage who have a background of perforated diverticulitis, a communicating tract should be suspected between the abdomen and the thigh. Once evidenced on the CT/MRI, Hartmann’s procedure should be offered earlier after careful consideration, even without a demonstrable intra-abdominal collection.


Reactive lesions of soft tissue are common oral lesions that are usually non-neoplastic growths without pain; however, they can interfere with oral hygiene and plaque control, and if present in the anterior segment, they can cause esthetics problems. In this case, there was a reactive lesion at the gingiva of the left maxillary central incisor area. The particular consideration about this case was the recurrence of a peripheral ossifying fibroma as a pyogenic granuloma, indicating the association between reactive gingival lesions. The history of several recurrences due to incomplete removal shows the importance of complete excision for preventing recurrence. Therefore, the lesion was completely excised, and the mucogingival defect was successfully augmented with periodontal plastic surgery. Dentists during routine examinations might face peripheral reactive lesions of soft tissues. Since oral mucosa is under the continual influence of different irritants, it exhibits a range of disorders, injuries, inflammation, and neoplastic conditions.Reactive lesions are common on the gingiva and tend to be not painful, without neoplastic growths, but often interfere with plaque control and cause patient discomfort. Most of these lesions are discovered between the fifth and sixth decades of life with a predilection for females. Histologic analysis is necessary for diagnosing these lesions because they are often clinically indistinguishable, but due to diagnosis, the recurrence rate might be different.The treatment of reactive lesions is surgical excision, which can be carried out by a scalpel blade, laser, or electrosurgery. A complete treatment should also consider determining the main etiology through a precise clinical evaluation and elimination of traumatic habits or any other etiological factors that led to the reactive lesion. Dental hygiene maintenance with professional follow-up care should reduce the recurrence prevalence for most types of gingival lesions.Since these lesions might be present for a long time, they might be traumatized and ulcerated. After treatment, the recurrence rate is 5–20% based on the diagnosis, complete removal, and the elimination of irritating factors related to their etiology.The classification of common gingival overgrowth lesions is as follows: pyogenic granuloma (PG), peripheral ossifying fibroma (POF), peripheral fibroma (PF), and peripheral giant cell granuloma (PGCG).These lesions are not regarded as neoplasms, and histopathological analysis can identify their diagnosis. They are considered reactive lesions and occur due to local irritants such as trauma, faulty restorations, dental plaque, calculus, and iatrogenic factors.Since these lesions’ clinical findings are similar, a histological analysis must confirm the definite diagnosis of the lesion.However, they are benign in nature; incomplete removal of the lesion or persistence of the local irritants leads to their tendency to recur. Hence, the treatment is surgical excision and complete removal to reduce the recurrence rate.Furthermore, the consequence of complete excision of the lesion might be a periodontal defect without keratinized tissue, with root exposure and esthetic problems.Thus, the ability of the surgeon to get a decent result involves augmentation and repair of the mucogingival defect and complete excision of the lesion. All of these can be performed during one surgical procedure with proper planning.This paper presents the management and treatment of a reactive lesion of soft tissue in the gingiva of the left maxillary central incisor with a history of several recurrence episodes due to incomplete removal, compelling us to perform a complete excision and consequently a free gingival graft to regenerate the excised tissue.A 52-year-old female patient referred to the Department of Periodontics at Tabriz University of Medical Sciences, complaining of esthetic problems due to a soft tissue lesion located on the gingiva of left maxillary central incisor, without pain, with a history of almost seven years and several attempts of incomplete removal and recurrence .The last biopsy had led to a diagnosis of a peripheral ossifying fibroma. Recurrence had occurred due to incomplete removal of the lesion. Intraoral examination revealed a lesion measuring approximately 1×0.6×0.5 cm, which was nodular, exophytic, and sessile. The radiographic image showed normal structures and no related changes.The surgery was performed under local anesthesia with 2% lidocaine and epinephrine at a concentration of 1:100,000. The excision of the lesion was instituted with a #15C blade. The lesion and the involved periodontal tissue were removed, followed by thorough curettage of the underlying bone and root planing of the tooth .After the excision, the lesion was taken with fine tissue forceps and transferred to a specimen bottle containing 10% neutral buffered formalin with almost 20 times the sample’s volume. A biopsy data sheet was completed, and the specimen was delivered to the laboratory.A free gingival graft was harvested from the palate to place in the exposed area to regenerate the lost periodontal tissue due to the excision.A sterile aluminum foil template of the recipient site was made to be used as a pattern for harvesting the graft from the palate. The template was placed over the donor site, and a shallow incision was made around it perpendicular to the tissue with a #15 blade (#15 blade was selected due to its wider tip because we only needed the tip of the blade) .The second incision was made in several steps to ensure inserting the blade perpendicular to the first incision to achieve an equal thickness all over the graft.The graft consisted of epithelium and a thin layer of underlying connective tissue with a thickness of 1.5 mm.After the graft was separated, it was immediately transferred, the excess clot was removed from the recipient site, and the graft was positioned and firmly adapted with non-absorbable monofilament polypropylene 4-0 sutures .The recipient and the donor site were covered with a eugenol-free periodontal dressing  to comfort the patient and reduce postoperative complications .After providing postsurgical instructions, the following medications were prescribed:Amoxicillin 500 mg q8hr for one week, Ibuprofen 400 mg q6hr PRN, and 0.12% chlorhexidine mouthwash q12hr for two weeks.Ten days later, the sutures and periodontal dressing were removed at the first follow-up, and recall arrangement continued at 2 weeks and 1, 2, and 6 months after the first follow-up.The pathological analysis confirmed the reactive nature of the lesion and reported a highly vascular proliferation in connective tissue resembling granulation tissue, covered by hyperplastic stratified squamous epithelium, diagnosed as a “pyogenic granuloma.”Reactive lesions of soft tissue are common oral lesions that are clinically indistinguishable from one another; therefore, it is important to differentiate them using histological analysis. Also, gingival tissues in the anterior mandible and maxilla are the most affected areas that might cause esthetic problems. Complete removal of the lesion and the elimination of irritating factors and their etiology are necessary for reducing the recurrence rate.Gingival tissues are exposed to constant irritation of bacterial plaque and biofilm. Reactive lesions generally arise from the periodontal ligament and connective tissue; therefore, the gingiva is the most common reactive lesions site.There are few articles indicating an interrelationship between these lesions. For example, POF and PGCG are site-specific lesions arising from the periodontal ligament and share histological features, such as abnormal connective tissue proliferation with varying amounts of giant cells and focal osteogenesis. However, PGCG is composed of relatively more immature and loose components compared to POF. reported a case of PGCG with extensive osseous metaplasia or a hybrid PGCG-POF.This case raises the question of whether non-neoplastic proliferative lesions are part of a disease spectrum or whether some of these lesions are true hybrid lesions.Ogbureke et alFurthermore, there are few case reports indicating an interrelationship between PG and POF, similar to our case. recommended that PG and POF might be different stages of the same pathology. Sridhar et al also reported a case of PG in the mandibular anterior segment, which was surgically excised but recurred after a year as a POF in the same segment. The authors concluded that regular follow-up is very important to avoid recurrence. Singhal et al reported the recurrence of a POF as a PG within one week. This case emphasizes complete excision to prevent a recurrence.Prasad et alIn our case, unacceptable esthetic was the patient’s chief complaint; therefore, we prepared a dual-purpose treatment plan, which included excision of the lesion, along with the regeneration of the anterior defect caused by excisional biopsy. In this treatment, excisional biopsy and free gingival graft were performed during one surgical procedure.Since there was a history of several recurrences due to incomplete removal of the lesion in the past, complete removal and thorough curettage of the underlying bone were necessary for treatment success.In our case, after placing the graft, the defect resulting from the excisional biopsy was fully reconstructed. The patient was satisfied with the esthetic appearance and without any inflammation or bleeding on probing after six months of follow-up .Reactive lesions are common oral lesions, and the treatment for all the common reactive lesions includes surgical excision and complete removal to prevent a recurrence, but it might result in a mucogingival defect.Therefore, this case report highlights the augmentation and repair of the mucogingival defect and the lesion’s complete excision to achieve satisfactory results. All of these can be performed during one surgical procedure with proper planning.RC carried out the procedures; MF carried out the procedures and supervised the project; NF prepared the manuscript, searched the literature. and designed the figures. All authors have read and approved the final manuscript.The authors state no competing interests.The patient signed a consent form before surgery and publishing this report.


Ameloblastoma is a rare benign epithelial odontogenic neoplasm, but with great clinical implications, as despite its benignity and slow growth, most cases are locally aggressive with a significant recurrence rate. Histological, cellular, or molecular analyses of its pathogenesis have confirmed the complexity of this neoplasm. We present the case of a 20-year-old patient with a suggestive clinical and radiographic diagnosis of ameloblastoma. An incisional biopsy was obtained confirming the diagnosis of conventional ameloblastoma. Left hemimandibulectomy and plate reconstruction were performed. Histopathological analysis of the surgical specimen confirmed the conventional ameloblastoma with a plexiform pattern and significant areas of cystic degeneration and amyloid-like-like deposits. Additionally, a microarray was carried out with bioinformatic analysis for the enrichment, protein interaction, and determination of eight hub genes  related to their pathogenesis. The World Health Organization defines ameloblastoma as a benign intraosseous epithelial odontogenic neoplasm, characterized by progressive growth with the destruction of surrounding tissues and a tendency for local recurrence if it is not adequately removed. It was first reported by Cusack in 1827 and named by Ivy and Churchill in 1930 [An annual incidence of 0.5 cases per million inhabitants is estimated; this makes it one of the most common odontogenic tumors, representing approximately 11% of all odontogenic tumors, with a higher incidence between the fourth and fifth decade of life, without predilection for any gender .Approximately 80% of all ameloblastomas are located in the posterior region of the mandible, followed by the anterior mandibular region, and posterior and anterior maxillary segments. Involvement of the sinus and nasal cavity regions is rare but has been reported ,5,6,7.Clinically, it presents as an asymptomatic growth or volume increase accompanied by expansive bone destruction or even cortical perforation. It is common for its early detection to be conducted by imaging finding, observing multilocular or unilocular radiolucent lesions. When the tumor reaches large proportions, deformity, masticatory dysfunction, facial asymmetry, opening limitation, or airway obstruction are observed. These features mean the clinical diagnosis will be more predictable; however, at these stages, the prognosis and quality of life of the patient are reserved, since radical surgical intervention is the alternative to his treatment ,10,11,12Its etiology and pathogenesis are still a subject of research. It has been considered that the neoplastic transformation of dental lamina remains; enamel organ elements and epithelial lining of some odontogenic cysts or basal epithelial cells of the oral mucosa may be the histological component for their pathogenesis ,4,13. DeThe bioinformatics methods on microarray profile datasets represents an important opportunity and strategy to correlate the clinical and molecular features with the possible application of therapeutic and/or prognostic biomarkers of ameloblastoma. Through these assays, the existence of differential changes in inflammatory and immunological mechanisms, MAP kinases, and cell cycle deregulations in ameloblastoma has been determined ,17,18,19A 20-year-old male patient refers to facial asymmetry after 3 months of evolution with a volume increase in the middle and lower left facial third, with painful symptoms. Radiographically, a multilocular radiolucent area was observed in the body, and the entire ascending mandibular branch was associated with the retained third molar. Intraorally, expansion of the lingual and vestibular cortices was observed, as well as a polypoid projection in the left mandibular retromolar area .The presumptive diagnosis was a probable neoplasm or odontogenic cyst, mainly ameloblastoma versus an odontogenic keratocyst, respectively. An incisional biopsy was performed under local anesthesia, obtaining a specimen of 1.9 × 1.3 × 0.8 cm with irregular edges, a light brown color, and soft consistency. Histopathological analysis revealed an odontogenic neoplasm compatible with conventional ameloblastoma, that presented anastomosing cords organized in a plexiform pattern with lax centers with palisade basal cells immersed over a dense connective tissue stroma.Due to the above histopathological results and the clinical features, it was decided to perform surgical treatment for complete removal of the lesion with safety margins and a definitive histopathological study, for which the patient was sent to the Department of Maxillofacial Surgery of the Balbuena General Hospital to perform the surgical protocol. Through the tomographic study, the extension of the lesion was evaluated; the tumor extension was from the mandibular body at the level of the premolars to the ipsilateral condyle, and it was decided to perform a hemimandibulectomy. Customized cutting guides were made with safety margins of 1.5 cm. For its reconstruction, it was carried out using a designed PMMA 3D customized glenoid fossa with a titanium mandible condyle and a locked 14-hole reconstruction plate 2.4 system. With the previous tomographic study, stereolithography was built to fit the reconstruction plate and evaluate the position of the temporomandibular joint (TMJ) prosthesis. Under balanced general anesthesia, Risdom-type submandibular access was performed to expose the lesion, cutting guides were placed and a reciprocating saw cut was made considering the safety margin, the mandibular segment was extracted with the complete lesion, and a joint prosthesis with the plate was placed. The reconstruction plate was fixed with 3 14 mm locking screws. The surgical sample was sent to the Oral and Maxillofacial Pathology department of the National Autonomous University of Mexico for final study. The final histopathology report of the surgical specimen confirmed the diagnosis of conventional ameloblastoma with a plexiform pattern, presenting significant areas of cystic degeneration, and the presence of an acellular amyloid-like amorphous eosinophil material deposit, with capsule, preserved 0.8 cm from the anterior border and 1.5 cm from the posterior border .p < 0.05 as significant. A total of 540 genes were analyzed for the enrichment process by software DAVID 6.8, available online: https://david.ncifcrf.gov (accessed on 7 November 2022), obtaining the differentially expressed genes (DEG) through Gene ontology for biological process (BP), molecular function (MF), and cellular component (CC), and Kyoto Encyclopedia of Genes and Genome selecting only elements statistically significant with a p < 0.05 [http://string-db.org/ (accessed on 7 November 2022) [http://kmplot.com/analysis (accessed on 7 November 2022), adjusting the follow-up threshold to 60 months with head and neck squamous carcinoma of overall survival considering an HR > 1 and a p < 0.05 [During the surgical procedure, a representative fragment of the surgical specimen was obtained (1 × 1 × 2 cm approximately) for total RNA extraction by trizol reagent protocol  and microarray analysis. An unerupted third molar dental follicle from an 18-year-old patient extracted by orthodontic indication was used as a control. An amount of 2 μg of total RNA from the ameloblastoma and dental follicle were used for the synthesis of cDNA and to perform microarray on the H35K_06_17 AFFYMETRIX Chip, , the control cDNA was labeled with Alexa 555 and the cDNA ameloblastoma with Alexa 647, followed by mixing and hybridization with the Chip. The overregulated genes were selected through the GenArise software considering only genes with Z > 2 . Posterier 2022) . The intp < 0.05 , to deteCurrently, with 15 months of evolution, the patient shows adequate facial contour and symmetry. The control orthopantomography showed complete resection of the lesion, without data of its permanence or recurrence, and reconstruction material in adequate position and function, waiting for prosthetic rehabilitation .According to the World Health Organization in 2017, ameloblastomas are classified into (1) conventional ameloblastoma (previously known as “solid/multicystic ameloblastoma”) which includes patterns: follicular, plexiform, acanthomatous, acanthomatous histopathologic variants, granular cell, basal cell, and desmoplastic; (2) unicystic ameloblastoma with luminal, intraluminal, and mural variants; (3) extraosseous/peripheral ameloblastoma; and 4) malignant or metastatic ameloblastoma. Histologically, it is characterized by tumor nests that resemble the epithelial component of the enamel organ, columnar to cuboidal cells with hyperchromatic nuclei arranged in a palisading pattern with reverse polarity, and the central core is reminiscent of stellate reticulum  malignan,25,26.The clinical course of odontogenic tumors in most cases is indolent, that is, the patient becomes aware of their presence when he observes increases in volume that affect swallowing or phonation, or due to a radiological finding that presents an important affection of maxilla or mandible. When this occurs, the treatment alternative is surgical . SurgicaIdentification of clinical-surgical factors for recurrence, as well as the histological, cellular, and/or molecular understanding, could help in the construction of better clinical, prognostic, and follow-up decisions ,38,39,40Post-surgical follow-up of patients with hemimandibulectomies must determine the postoperative facial profile and contour in a clinical and radiographical approach along with the facial contour symmetry and occlusion analysis . By 15 mThe recurrence potential observed in ameloblastoma always makes one think about the best surgical alternative to use. Thanks to the selection of hemimandiulectomy at 15 months of follow-up, no recurrence is observed. The reconstruction through a bar and PMMA personalized glenoid cavity have shown an acceptable state. However, longer-term follow-up is necessary, to minimize side effects and aesthetic and functional complications.In this case report, it is the first time that a designed PMMA 3D customized glenoid fossa as part of the TMJ replacement systems has been reported, as well as the use of bioinformatic analysis of the gene microarray as a mechanism to understand the clinical and histopathology findings presented in this case. The histopathological analysis is essential not only for diagnosis and therapeutic planning but also as a prognostic factor. The detailed histological analysis of odontogenic tumors is mandatory, considering neoplastic parenchyma and stroma, as well as its relationship with adjacent structures through the surgical edges, which confirms that this analysis is a necessary tool. Using new analysis techniques such as gene microarrays, the information of thousands of genes can be obtained; it is the task of bioinformatics to put an order in these results. Thanks to new databases, bioinformatic estimation for the association of these results with new variables opens the door to innovative research. An interesting finding was the histological presence of amyloid-like deposits, which could correlate with the upregulation of the APP gene. In our opinion, the high-throughput analysis as microarray validates the histopathological findings, increasing our internal validity; however, if we want extrapolation, it is necessary for another research design in clinical investigations, maybe a cases and controls study.


Autotransplantation of teeth as an alternative to dental implantation is characterized by short healing periods, preservation of aesthetics and propriosensitivity in the area of the transplanted tooth and the possibility of its orthodontic movement. This clinical case describes a successful delayed autotransplantation of the third maxilla molar (2.8.) with the completed root formation into the socket of a previously removed tooth 1.6. with perforation in the maxillary sinus area on the right and the presence of signs of chronic inflammation. Long-term observations after 30 months demonstrate favorable healing with restoration of dentoalveolar attachment in the area of the transplanted tooth, relief of inflammatory process in the area of the maxillary sinus with restoration of the cortical plate. Key words:CBCT, tooth transplantation, dental autotansplantation, wisdom teeth. Despite the fact that today dental implantation is one of the priority methods of treating the absence of teeth, the possibility of its use depends on a number of conditions. One of these conditions is the state of the bone tissue in the area of the planned intervention. Bone tissue deficiency can be caused by a plenty of reasons: traumatic tooth extraction, extensive inflammatory process, close location of the maxillary sinuses, etc.Obviously, performing bone regeneration techniques increases the risk of possible complications , as wellAn alternative method for the treatment of single defects of the dentition is considered to be autotransplantation of the tooth, which becomes a predicTable option for the restoration of chewing and aesthetic function ,3. Up toThis article describes the method of autotransplantation of a tooth in the case when dental implantation would be associated with a longer and more complex treatment.-Patient informationA 35-year-old patient came to the clinic 7 days after tooth extraction 1.6, complaining about the presence of communication of the oral cavity with the maxillary sinus, difficulty in eating. There were neither relevant medical problems or allergies. The family history was unremarkable.-Clinical findings While the examining the oral cavity, the socket of the extracted tooth 1.6. filled with a fibrin clot was determined. The sutures were well-maintained, covered with a soft plaque. Oral-nasal and nasal-oral tests were positive. When probing the distal surface of the tooth 1.5. a periodontal pocket with a depth of 10 mm was determine. The mucous membrane surrounding the tooth had pale pink color, without pathological changes and signs of inflammation. There was no discharge from the socket. There were no clinical signs of acute maxillary sinusitis.-Diagnostic assessmentThe analysis of cone-beam computed tomography (CBCT) revealed a local vertical loss of bone tissue along the distal surface of the tooth 1.5, the absence of a part of the vestibular cortical plate in the area of the socket of the extracted tooth 1.6. and the bottom of the maxillary sinus on the right, as well as signs of parietal thickening of the mucous membrane in the area of the bottom of the maxillary sinus , which is not of functional value, the patient chose a treatment plan that includes autotransplantation of the tooth.Surgical intervention10 days after extracting 1.6 tooth, patient was prescribed with amoxicillin/clavulanic acid 875mg/125mg 1hour prior to surgery. Under local anaesthesia with 4% articaine hydrochloride plus 1:100,000 epinephrine, sulcular incisions were made in the teeth area 1.5, 1.7, vertical incision in the tooth area 1.5. The muco-periosteal flap was elevated from the vestibular surface to the borders of the vestibular cortical plate bone defect , the maxillary sinus was located in the molar area  and accoImplantation is often impossible or involves great risks at each stage of treatment: the presence of chronic inflammation, perforation of the sinus, in the process of removal, traumatization of the vestibular wall of the alveolar process. Referring to Esposito M, Felice P., Worthington HV, the probability of complications after sinus lifting increases by 4.77%. Often tAutotransplantation of teeth also has a number of limitations. Most adult donor teeth are having formed root, which means that patients will need endodontic root canal treatment after transplantation. In addition, one of the most common complications of autotransplantation is root resorption and attachment loss . To prevIn presented clinical case, at the time of the patient’s treatment, significant bone atrophy and oroantral fistula were visualized, the initial conditions for the restoration by implantation were rather unfavorable. The plan of implantological treatment would include several surgical stages: elimination of the oroantral fistula, open sinus lifting using bone-plastic materials, in order to eliminate vertical bone loss in the distal part of a tooth 1.5, implantation itself, then installing the gingiva former, then permanent prosthetics. Summarizing: treatment would take at least one year and would involve certain risks at each stage of treatment. AutotraAutotransplantation is considered the most predicTable option for replacing a lost tooth, as no plastic bone surgery needed. Studies have shown survival rates of 84%-94% of autotransplanted teeth .Like any method of dental replacement, autotransplantation has its advantages and disadvantages. With a properly performed operation, the transplanted tooth functions as a normal one and can be even moved orthodontically , and alsThe clinical case demonstrates the possibilities of autotransplantation.This operation significantly reduces the duration of patient’s rehabilitation process, while ensuring minimal invasiveness: a gentle attitude to soft tissues, no need for bone plastic surgery, the possibility to use it as the base for orthopedic construction, also the tooth can be moved orthodontically.


Malignant melanotic nerve sheath tumor (MMNST), formerly called melanotic schwannoma, is a rare tumor of neural crest derivation which most frequently arises from the region of spinal or autonomic nerves near the midline. Recent studies have reported malignant behavior of MMNST, and there still has no standard management guidelines. Intra-abdominal MMNST, which has never been reviewed as an entity, is even rarer. In this study, we present a rare case of a cystic MMNST arising from the para-aortic region and mimicking an intra-abdominal gastrointestinal stromal tumor (GIST), and review the literature regarding MMNSTs located in the abdominal cavity.A 59-year-old female was incidentally found a tumor located in the left para-aortic area by non-contrast computed tomography. A Magnetic Resonance Imaging showed a cystic mass originated from the inferior mesenteric artery (IMA) territory. A GIST was initially diagnosed. The tumor was resected en bloc by laparoscopic surgery and was found between mesocolon and Gerota’s fascia with blood supply of IMA. Grossly, dark brown materials were noted at the inner surface of the cystic wall. Microscopically, the tumor cells were melanin-containing, and no psammomatous bodies were present. Immunohistochemically, the tumor showed positivity for MART1, HMB45, collagen IV, and SOX10, and negativity for AE1/AE3. MMNST was favored over malignant melanoma, since the tumor was located near ganglia and had cells with less atypical cytology and a low mitotic rate, and subsequent adjuvant radiotherapy was performed. The patient was alive with no evidence of recurrent or metastatic disease 11 months after radiotherapy.Our review of abdominal MMNST cases showed a female predominance, with an average age of 54.8 years, and a trend toward being a larger tumor showing cystic or necrotic changes. Local recurrence and metastasis rate were reviewed, and both showed a low rate. Diagnosis of MMNST should combine all the available findings, and complete excision of the tumor should be performed, followed by long-term patient monitoring. Malignant melanotic nerve sheath tumor (MMNST), also called melanotic schwannoma, is a rare tumor of neural crest derivation composed of variably melanin-producing Schwann cells , 2. MMNSMMNST still has no standard treatment guidelines, and complete excision of the tumor is the most common approach. In the present study, we present a rare case of a cystic MMNST arising from the para-aortic region and mimicking an intra-abdominal gastrointestinal stromal tumor (GIST). We also discuss the radiologic and histologic features, and the literature regarding MMNSTs located in the abdominal cavity is reviewed.A 59-year-old female was referred for a tumor located in the left para-aortic area, close to the pancreatic tail and left renal hilum, found incidentally by non-contrast computed tomography (CT) during a urolithiasis survey. This patient had a past history of hypertension, but it was under control. She was asymptomatic, without any abnormal physical examination findings.−3 mm2/s. The cystic part showed hypointensity on T1WI images  further revealed a 4.7 cm cystic mass at the para-aortic region abutting to the 3rd and 4th portion of the duodenum . The pigment was positive for melanin staining and negative for iron staining. Immunohistochemically, the tumor showed positivity for MART1, HMB45, collagen IV, and SOX10, and negativity for AE1/AE3  and 11 were female (68.75%). Torres-Mora et al. reported 18 male and 22 female patients in 40 cases of all locations, whereas other previous studies reported no sex predilection 5% and 11. The tumWe also assessed the cystic or necrotic changes that occurred in the abdominal MMNSTs. Overall, 14 cases provided structural descriptions, and cystic or necrotic changes were mostly observed in larger sized tumors  but less frequently noted in the smaller sized tumors . All patients underwent surgical removal of the tumor, and no cases had incomplete surgical resection. Follow-up was available in 12 cases, ranging from 7 months to 60 months. The recurrence or metastatic rate were both reported for 1/12 8%) of these patients and revealed a relatively lower rate than in recent studies , 25. The% of thesMMNST, as updated and defined by the current fifth edition of the WHO classification of tumors of soft tissue and bone , was forPRKAR1A gene deficit [The etiology of MMNST is still unknown. Theories have included melanomatous transformation of neoplastic Schwann cells, phagocytosis of melanin by Schwann cells, and the presence of two different neoplastic populations of proliferating melanocytes and Schwann cells . An asso deficit . However deficit , 4.Abdominal MMNST often presents with nonspecific abdominal symptoms or is discovered as an incidental finding. GISTs are the most common mesenchymal tumors ; therefoGrossly, MMNST presents as a dark brown or black tumor, and it sometimes presents with hemorrhagic components, cyst formation, or necrosis . The tumPRKAR1A gene is seen in most MMNSTs [BRAF V600E. Negativity for this test would also indicate MMNST [Genetically, mutation of the t MMNSTs . The prete MMNST , 33.Gross total excision is widely accepted as the primary treatment for MMNSTs. The local recurrence rate and metastatic rate remain controversial, with < 15% stated in the past and 42% reported in more recent studies . AdjuvanIn this study, we reported an incidentally found intra-abdominal MMNST and reviewed the features of this entity in a total of 18 cases. This study had some limitations. MMNST is an extremely rare entity; therefore, the small sample size, inconsistent treatment protocols, and incomplete follow-up restricted the results of our study. Further evaluation of long-term follow-up and treatment are needed.Intra-abdominal MMNST is a rare entity. We presented a case with an initial diagnosis of GIST that was subsequently identified as an MMNST located at the para-aortic area. Our review of abdominal MMNST cases showed a female predominance, with an average age of 54.8 years, and a trend toward being a larger tumor showing cystic or necrotic changes. Local recurrence and metastasis rate were reviewed, and both showed a low rate. Diagnosis of MMNST should combine all the available findings, and complete excision of the tumor should be performed, followed by long-term patient monitoring.


Most patients with splenosis have no clinical symptoms and do not need intervention. Hematospermia and testicular pain occurred in this patient, which was considered to be related to the huge pelvic implantation of the spleen, which was relatively rare in clinical practice, so we hereby report this case.A 28-year-old male patient with a history of splenectomy was admitted to the Urology Department of the Second Affiliated Hospital of Anhui Medical University with the chief complaint of "Hematospermia for 1 month and testicular pain for 2 days". Preoperative imaging examination indicated pelvic mass. Combined with the patient's history of splenectomy for splenic rupture in childhood, the possibility of pelvic spleen implantation was considered. Laparoscopic pelvic exploration was performed. During the operation, multiple grayish-brown nodular tissues were observed in the space between the posterior bladder and rectum, and a lobulated grayish-brown mass with a diameter of about 9 cm was observed in the posterior upper part of the prostate gland and seminal vesicle at the pelvic floor. Two nodular tissues were removed intraoperatively and sent for quick frozen pathology, which was reported as spleen tissue. Further resection of the huge mass was performed, and the postoperative pathological results were consistent with the diagnosis of splenosis.We report a rare case of splenosis presenting with hemospermia and testicular pain. Splenosis is a common benign lesion, which is usually found incidentally. After splenic rupture or splenectomy, the splenic fragments or splenic cells spread to the abdominal cavity or adjacent organ tissues. These cells or tissues are implanted, and they receive blood supply from adjacent tissues and gradually differentiate into mature splenic tissues –3. Due tThe 28-year-old male patient was admitted to the Department of Urology of the Second Affiliated Hospital of Anhui Medical University on December 24, 2020 due to "hemospermia for 1 month and testicular pain for 2 days". The patient began to ejaculate with occasional bloody semen 1 month before admission. Right testicular pain started two days before admission. The patient underwent open splenectomy due to rupture of the spleen 20 years ago, and no other past medical history was found. Color ultrasonography of prostate, seminal vesicle, spermatic cord, testis and epididymis indicated enlargement of right seminal vesicle and enhanced echo. The left seminal vesicle gland was normal, and the bilateral ejaculatory duct was not significantly dilated. There was a 74 * 53 mm cystic mass with poor echo posterior to the right side of the prostate. The right epididymis was enlarged and the echo was decreased. There was a dense speckled echo in the right epididymis with abundant blood flow. Pelvic MRI plain scan + DWI + enhancement indicated abnormal signal above the posterior to the seminal vesicle, which was closely related to the seminal vesicle, considering the origin of the seminal vesicle and the possibility of neoplastic lesions. The right seminal vesicle gland was slightly enlarged and the T2W1 signal decreased (Fig. Although the blood supply of most implanted spleen is limited, the pelvic mass of this patient was large, which did not rule out the possibility of abundant blood supply. Preoperative blood preparation was actively conducted. The patient had a history of open surgery, but considering that it has been 20 years since the last operation, the intraperitoneal adhesions have been absorbed and the operation was mainly performed on the upper abdomen at that time, and the adhesions in the lower abdominal organs may not be serious. In addition, laparoscopic technique has the advantages of clear surgical field, less bleeding, less trauma to patients, less postoperative complications and quick postoperative recovery, so we choose to perform this operation under laparoscopy . It was Splenosis occurs after rupture of the spleen or splenectomy , and itsHemospermia is usually caused by inflammation, the most common is seminal vesiculitis, but can also be caused by other reasons such as obstruction or cysts, stones, tumors, etc. . The repBased on this rare case, we made a series of advice, which are summarized as follows: First, the establishment of a good follow-up mechanism for patients after splenectomy or rupture of the spleen is not only helpful to observe the survival of the transplanted spleen, but also to monitor the growth and development of the ecotopic implanted spleen that may cause clinical symptoms . Second,For patients with a history of splenic rupture or splenectomy, urologists should pay attention to the individualized plan and comprehensively consider all factors in the diagnosis and treatment of patients, and do not ignore any small link. Especially for patients with abdominal or retroperitoneal masses, attention should be paid to the possibility of splenosis, and we should carefully identify the tumor and avoid excessive treatment.


A dermoid cyst is a benign congenital lesion of ectodermal origin that can arise in any region of the body, though occurrence is rare. A young girl aged 2 years 4 months was referred to our hospital because of a painless mass in the floor of the mouth. Intraoral examination findings revealed a painless movable elastic soft mass on the floor of the mouth measuring approximately 15 mm in diameter. Magnetic resonance imaging indicated a cystic lesion, with low signal intensity shown in T1-weighted and extremely high signal intensity in T2-weighted images. These clinical findings indicated the presence of a dermoid cyst and removal was planned. Under general anesthesia with nasal intubation, surgical removal was performed through an incision on the floor of the mouth. Blunt dissection exposed the integrity of the cyst capsule, which was weakly attached to adjacent tissue. The excised mass was 19 mm × 14 mm × 11 mm in size. Histological examination findings confirmed a diagnosis of dermoid cyst. The operation was successfully completed without any complications and the postoperative course was good. It is important to properly evaluate cysts in children and provide proper treatment with appropriate timing. A dermoid cyst is a benign congenital lesion with an ectodermal origin that can arise in any region of the body, though occurrence is rare ,2,3. ThiDermoid cysts are often misdiagnosed as an inflammatory pseudocyst (ranula) or lymphatic malformation . ClinicaMost dermoid cysts in the floor of the oral cavity are diagnosed between the ages of 10 and 30 years . Occurre® (triclofos sodium) as a sedative agent, an internal magnetic resonance imaging (MRI) examination was performed, which revealed a cystic lesion with low signal intensity in T1-weighted images and extremely high intensity in T2-weighted images  or lymphatic malformation . When thStandard treatment for a dermoid cyst on the floor of the mouth is surgical removal ,6. BasedRecurrence of a dermoid cyst or malignant transformation to a squamous cell carcinoma is extremely rare, and a postoperative infection is also unlikely to occur ,19. In tIncreased salivation, speech disturbance, dyspnea, and difficulties with eating, swallowing, and breathing may be present in a patient with a dermoid cyst, which are dependent on its size. Early treatment for removal is generally required, especially when the airway is compromised or swallowing difficulties are noted. When a cyst is noted in a child patient, proper evaluation and treatment at an appropriate time are essential.


Melanotic schwannoma is a melanin producing nerve sheath tumors. Rarely, it can be associated with psammoma bodies, called psammomatous melanotic schwannoma. Psammomatous melanotic schwannomas are associated in up to 10% of the cases with Carney’s syndrome. The rarity of the lesion, which may present at different localizations create difficulty in placing a correct initial diagnosis. Definitive diagnosis is made after complete tumor excision and pathomorphological evaluation. The prognosis depends on the anatomical localization, local invasion and presence of a high mitotic index. The main pathomorphological differential diagnosis includes schwannomas and other melanin producing tumors as melanoma.We present a case of an 11-year-old female with cystic lesion adjacent to right adrenal gland, mimicking adrenal cyst. Ultrasound guided biopsy was undertaken due to the cystic appearance of the formation and the lack of certain diagnosis from the non-invasive diagnostic tests. No signs of cellular and nuclear atypism were observed. The diagnosis of benign endothelial cyst with spontaneous hemorrhage was suggested. The patient underwent transabdominal laparoscopic adrenalectomy en-bloc with the cyst to prevent spillage of the cyst content due to the intimate adhesion of the lesion to the adrenal gland and vena cava inferior. Pathomorphological examination revealed malignant psammomatous melanotic schwannoma. The adrenal gland was intact with no tumor infiltration. The patient was followed up on the 1st and 2nd month afterwards the surgery by MRI with no signs of local recurrence and postoperative complications.Psammomatous melanotic schwannoma near adrenal gland are rare and present difficulty with exact preoperative diagnosis. Complete resection should always be provided. Laparoscopic surgery is feasible if radical excision is not compromised. Long-term follow-up and Carney’s syndrome surveillance after complete excision are recommended especially in young patients. Melanotic schwannoma (MS) is a melanin-producing nerve sheath tumor . Up to 5An 11-year-old female patient presented to the pediatric department with complaints of nausea, vomiting, and right lumbar pain lasting for 1 week, without previous symptoms and no history of trauma.2 (above 95th percentile on the CDC (Centers for Disease Control and Prevention) age- and sex-matched BMI growth charts), active striae and acanthosis on the inner thighs; Tanner pubertal stage 4+, with menarche starting 6 months prior the admission. The blood pressure was measured routinely and was within normal range.On physical examination, the following was noted: The weight was 82 kg and body mass index (BMI) of 27.0 kg/mThe initial laboratory hormonal analysis and tumor markers aimed to identify hormonal activity by the tumor or possible malignancy Table . The resThe computer tomography images showed a well-defined capsulated retroperitoneal extrarenal oval lesion with heterogeneous cystic structure measuring 7.5 × 8.5 cm, native HU 5 to 95 without change after contrast enhancement Fig. .Fig. 1CoUltrasound guided fine-needle aspiration biopsy was performed and a pig-tail drainage was placed due to the cystic appearance of the formation and the lack of certain diagnosis from the non-invasive diagnostic test. Cytological examination revealed a monomorphic population of cells with loose-textured cytoplasm and central lymphocytic-like cores with small nucleoli around a multitude of erythrocytes. No signs of cellular and nuclear atypism were observed. The drain discharged 80 ml of hemorrhagic fluid in the first 24 h and after being inactive in the next 24 h it was removed. The diagnosis of benign endothelial cyst with spontaneous hemorrhage was suggested due to imaging data suggesting a cystic lesion, the hemorrhagic characteristics of the drained fluid and the cytology analysis. The patient was observed for two days and discharged with no complains, normal vital signs and no significant change in the blood cell count. The hormonal status was to be evaluated on follow-up.2 was observed. Striae and inner thighs acanthosis remained. Additional hormonal laboratory tests ruled out adrenal pathology  were performed and demonstrated decrease of the cyst dimensions to 52 × 50 mm , with varying amount of melanin pigment in single and groups of cells. Connective tissue stroma separated tumor cell aggregates with an abundance of psammoma bodies  and foci of necrosis and hemorrhage in the tumor parenchyma. The adrenal gland was intact with no tumor infiltration Fig. .Fig. 5A The markers S100 protein, melan-A and melanosome clone HMB45 had high-intensity expression in the tumor. Individual cells expressed inhibin alpha, Chromogranin A and Ki 67 to about 10 to 15%  are hypointense on T1-weighted and hyperintense on T2-weighted sequences, opposite to melanotic Schwannomas which appear hyperintense on T1-weighted and hypointense on T2-weighted sequences based on the paramagnetic effects of their melanin-containing composition . Khoo etUp to date, there is no standard protocol for management due to rarity of this tumor. Surgery is the procedure of choice. When psammomatous melanotic schwannoma is suspected, biopsy of the tumor is not recommended because of risk for seeding of the tumor, hemorrhage or infection. In our case, due to uncharacteristic MRI and CT appearance, fine needle aspiration and drainage attempt was performed.Laparoscopy is the current established approach for treating retroperitoneal and especially adrenal lesions due to lower postoperative pain and hospital stay, complication rate and comparable operative results . ClinicaSchwannomas are neoplasms of Schwann cell origin. Although most schwannomas demonstrate classic histology, morphologic variations are occasionally encountered, such as cellular, plexiform, and melanotic schwannomas. The latter are characterized with epithelioid cells with variably sized nuclei and marked accumulation of melanin in neoplastic cells and associated melanophages. The main differential diagnosis is with other melanin-producing neoplasms, in particular melanoma. MS may occur in two forms, depending on the presence of psammoma bodies at histological examination . Carney The absence of any established post-treatment protocols underlines the necessity of long-term follow-up, especially in patients with proven aggressive histopathological findings at biopsy or resection . Steins Melanotic schwannomas are rare. Exact preoperative diagnosis is difficult to achieve and they might mimic pathologies of surrounding organs or structures because of their atypical presentation. The presented patient demonstrates a rare occurrence of malignant melanotic schwannoma initially recognized as cystic formation in the adrenal gland region. Laparoscopic surgery is feasible if radical excision is not compromised. High importance is established for the proper pathomorphological differentiation between melanotic schwannomas, melanoma, and pigmented neurofibromas due to their different biological behavior and treatment protocols. Long-term follow-up and Carney’s syndrome surveillance after complete excision are recommended especially in young patients.


Frontiers, a series of case reports describe the clinical courses of unique thoracic pathologies and provide valuable insight into how we can approach diagnosis and treatment of unusual cases.Rare thoracic pathologies and unusual presentations of more common ones can cause diagnostic and treatment dilemmas that may require a more tailored and multidisciplinary approach. In some cases, a definite preoperative diagnosis cannot be made, and the standard treatments are not as established as they are in common thoracic pathologies. In this issue of Frontiers describe unusual presentations of very rare pathologies, benign and malignant. In Xiang's et al. report, after a video-assisted thoracoscopic surgery (VATS) resection of a posterior mediastinal mass presumed to be a schwannoma, final pathology identified epithelioid angiosarcoma arising from a schwannoma. This represents an already-rare malignancy arising in a very unusual location, yet successfully managed with surgery alone. Zhi et al. describe a similar situation, in which an anterior mediastinal tumor, initially thought to be a teratoma, was pathologically diagnosed as an epithelioid hemangioendothelioma. Contrary to a teratoma, diagnosis of this rare angiogenic tumor necessitated adjuvant therapy and a rigorous follow-up schedule, without which the patient's recurrence 6 years later may have gone undetected and untreated.Some thoracic pathologies, especially those that are very uncommon, can cause misdiagnoses preoperatively or elude diagnosis altogether until pathologic examination can be performed. Most of the reports in this issue of Zhang et al. report the first case of a pulmonary benign metastasizing adenomyoma. This mass is thought to be a benign metastasis from uterine adenomyoma, and the authors reasonably draw attention to the patient's personal history of uterine leiomyoma and adenomyoma as risk factors. Six months after resection, the patient had no signs of recurrence; perhaps the patient's history imparts a lifetime risk of developing benign metastasis, which only long-term surveillance could tell. In the report by Hu et al., a mass thought initially to be Castleman's disease or a thymoma was found upon intraoperative inspection to be a left innominate vein aneurysm. The authors demonstrate that VATS is a viable approach for management of mediastinal venous aneurysms. If nothing else, these case reports are prime examples of why it is important to always keep a broad differential when working up thoracic masses.Identification of a rare pathology does not always imply malignancy, however. Cariboni et al. report on the radical resection of a locally advanced paravertebral undifferentiated pleomorphic sarcoma with involvement of the ninth thoracic vertebra and the aortic wall. The treatment team opted for a surgery-first approach, and the combined efforts of thoracic, vascular, and spine surgeons resulted in a successful two-stage R0 resection. At 22 months postoperatively, the patient remained without evidence of disease, representing a favorable outcome made possible by a carefully crafted, multidisciplinary preoperative plan. Yue et al. describe the management of a pulmonary primitive neuroendodermal tumor, a very rare malignancy found in an equally rare location. The size of the tumor−12 × 11 cm—necessitated pneumonectomy, which the patient tolerated well. Long-term follow up would help determine if radical resection alone can provide equivalent survival compared to surgery plus adjuvant therapy. In some cases, local recurrences are unavoidable though, despite the successful completion of a radical resection. This was the case described by Rastrelli et al., in which the patient had a rapidly growing radiation-induced undifferentiated sarcoma of the thoracic wall. In order to obtain negative margins, resection of the mass resulted in a thoracic wall defect that required extensive reconstruction to repair. Unfortunately, the patient developed an aggressive local recurrence and ultimately passed away. All of these cases highlight the importance of clear and open discussions weighing the risks and benefits of radical resection, including the potential for recurrence.There are some circumstances, such as cases involving very large or locally advanced malignancies, in which radical resection offers the best chance of cure. Cases such as these are ideally handled in a multidisciplinary fashion and sometimes involve coordination with other surgical specialties other than thoracic surgery . CaribonSignore et al. describe an uncommon presentation of a very common pathology: adenocarcinoma of the lung. After being placed on veno-venous ECMO for acute respiratory failure refractory to mechanical ventilatory support, the patient is found to have a T4N0M0 mass and undergoes a right inferior bilobectomy. Resection immediately improved the patient's oxygenation, indicating that the tumor had been causing a severe pulmonary shunt, and he was weaned off ECMO 3 days later. Although Stage IIIA non-small cell carcinoma is most often managed with neoadjuvant therapy followed by surgery, the patient's clinical status superseded the traditional standard of care. Although the patient recurred and ultimately died 1 year after surgery, the tailored approach to his care gained him a year of life.The use of extracorporeal membrane oxygenation (ECMO) support for patients with advanced thoracic malignancies has been documented but is not a common practice . In the Approaching rare thoracic pathologies and unconventional presentations can pose challenges for providers, whether due to the dearth of treatment recommendations or the extent of surgery that may be required to achieve cure. The reports in this issue focus on these unique clinical scenarios and will hopefully provide guidance to providers who are managing similarly challenging cases.Both authors listed have made a substantial, direct, and intellectual contribution to the work and approved it for publication.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. 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