It is known that specimen collection followed by histopathological workup is the core of evidence-based medical therapy of musculoskeletal tumors. There exist many controversies about how a biopsy should be performed. While some centers recommend minimal invasive biopsy procedures, mostly the core needle biopsy (CNB), others prefer the incisional biopsy.This study aimed to determine the accuracy of incisional biopsy for malignant tumors in the musculoskeletal system. Moreover, advantages and disadvantages to other biopsy methods are discussed.This retrospective, single-center study about 844 incisional biopsies  analysis the diagnostic accuracy of 332 malignant tumors, concerning the final histopathological result. In addition, surgical complications are analyzed to find the best way to plan and treat patients timely and correct. Secondary endpoints are the patients age, the pure operation time, as well as the type of tumor, and the subsequent therapy.In summary, incisional biopsy corresponded a sensitivity of 100% for malignancy in 844 incisional biopsies and a specificity of 97.6% in 332 malignant tumors, but it features greater operative expense (incision/suture 23.5 min) and the risk of general anesthesia.The method of biopsy should be tailored to the individual patient and the experience of the center performing the procedure. The biopsy is the critical diagnostic step for the appropriate treatment of all cancerous diseases. It is especially challenging in bone and subfascial soft tissue. Sarcomas are malignant tumors of mesenchymal cells, and they are generally distinguished in soft tissue sarcoma (STS) and bone sarcoma (BS). STS and BS are rare cancers in adults . BecauseThe purpose of this retrospective, single-center study is to investigate the diagnostic precision of incisional biopsy for STS, BS, and metastasis as well as lymphoma located in bone or soft tissue.The study had a retrospective, single-center design. The data collection and analysis were anonymous and in accordance with local state law (§17 HmbKG). Institutional Review Board approval was given. All patients were surgically treated from 2011 to 2021 at a university cancer center (UCC). A total of 844 incisional biopsies performed at our institute. To determine the diagnostic accuracy by means of sensitivity and specificity, we decided to evaluate the malignant histopathological results. In these cases, subsequent resection or oncologic system therapy was performed.Demographic clinical, radiological, and histopathological data were retrieved from the patients’ electronic medical chart. In most cases, radiological imaging was performed by residential radiologists and subsequently, based on the imaging diagnostics, the presentation took place in our musculoskeletal orthopedic center. In some cases, the imaging was performed directly in the UCC.All incisional biopsies were performed under general anesthesia. After the procedure, patients were observed for at least 2–4 h to ensure the absence of immediate complications, such as dizziness, bleeding, vomiting, or neurovascular injury.Adult patients with malignant tumors in the radiologist’s report and the histopathologic report was collected from the electronic medical charts.Patients under 18 years of age were excluded in this single-center study, because the UCC treats only adults. Furthermore, all patients with biopsies or incomplete tumor resections, which were performed in other health care institutions and only then referred to the UCC.All surgical specimen was analyzed in the pathology department of our institution. In cases of doubt, a reference pathology was contacted.Statistical analysis: The data was analyzed with GraphPad 9 Software . Parametric distribution was tested by the Smirnov-Kolmogorov test. Different cohorts were compared to with ANOVA. A confidence interval of 95% was chosen. Mean values are followed by standard deviation in (±).All included patients presented to the UCC were based on imaging suspicion of a malignant mass in soft or bone tissue. After reviewing the previous findings, taking a detailed medical history, examining the patient clinically and, if necessary, performing additional diagnostic tests, we decided to perform a diagnostic surgical incisional biopsy. The mean time between the initial patient presentation and incisional biopsy was 8.8 days SD: 8.4). 332 individual patients were included to our study, 129 (39.2%) were female and 203 (60.8%) male patients. In 332 biopsies performed, histopathologic workup diagnosed 142 STSs, 34 BTs, 69 lymphomas, and 87 metastases  min. The average cut/suture time of the bone sarcomas was 27.2 (±17.1) min. For lymphomas, it took on average 21.2 (±8.2) min from incision to suture. Finally, the average surgery-only time for proven metastases was 24.3 min with a standard deviation of 13 min Fig. .Fig. 6AvAll incisional biopsies were performed under general anesthesia without complications. The mean time between the incisional biopsy and the final histopathological finding was 10.6 days. Only 6 of the 332 biopsies needed to be repeated because the pathohistological analysis could not produce a valid result. In the remaining 326 tumors, the incisional biopsy yielded a valid diagnosis. In 142 cases, a primary oncological resection followed. In 70 cases, the patients received adjuvant radiotherapy or chemotherapy. Mainly, because of progressive metastatic cancerous disease. All therapeutic decisions for malignant tumors were made by the interdisciplinary tumor board and for each patient individually.The primary tumor, which was surgically removed was histopathological analyzed and there was a 100% concordance to the histopathologic diagnosis from the biopsies. All open biopsies were found to be highly accurate with a sensitivity of 97.6%. In the 2.4% cases of a frustrated or inadequate biopsy, re-biopsy was performed with evidence of malignancy. In two cases, a complication in the sense of a hematoma requiring surgical treatment was seen postoperatively. In these cases, the hematoma was evacuated promptly.All patients present with unclear masses both in the ambulant setting as well as in specialized musculoskeletal centers. After appropriate imaging and finding of a suspected diagnosis, a biopsy is often necessary for confirmation to plan the further procedure. Prompt referral for further diagnostics is particularly important if a malignant disease is the possible cause of the mass. Although many biopsy techniques have been described, there is currently no standardized procedure. For example, Errani et al. described the current concepts in the biopsy of musculoskeletal tumors, which primarily include fine needle aspiration, core needle biopsy, or incisional biopsy, and came to the conclusion that there is still no clarity about the optimal biopsy acquisition .Due to the low risk of complications and the lower costs, they recommended CNB to be preferred, but if this remains without a result, an incisional biopsy should be performed .Once more, this shows the discordance about the validation of the different methods, especially CNB versus incisional biopsy. While some centers prefer CNB as the method of choice and publish good results in the available literature, primarily IB is used in our center with also very good results. The division of the histopathological findings into the individual tumor types makes it clear that IB shows the same sensitivity not only in soft tissue, but also in bone tissue, and has a high diagnostic confidence in solid tumors as well as in the malignancies of the hematopoietic system and metastases. From our point of view, what is important is the uncomplicated acquisition of a biopsy without a high risk of complications, the rapid availability, and the correct diagnosis. The purpose of this study was to investigate the accuracy of incisional biopsy in cases of existing suspicion of a malignant tumor of the musculoskeletal system. Especially in the case of malignant tumors, the most precise, timely, and complication-free biopsy possible is of elementary importance for the correct diagnosis and appropriate planning of further therapy .Recently, Birgin et al. reported in a systematic review about diagnosing the correct soft-tissue-tumor type. The conclusion of their meta-analysis demonstrated that CNB is not inferior to IB. Sensitivity was calculated as 97% in the CNB group and 96 in the IB group for malignancy. For soft-tissue-tumors: CNB 88% and IB 93%. It is debatable whether CNB may provide higher diagnostic confidence in bone sarcoma due to its fixed position in solid tissue, whereas in soft tissue sarcomas, the displaceability of the mass, patient positioning, or the biopsy itself may significantly reduce sensitivity. In this case, open biopsy is clearly superior, as it is possible for the experienced surgeon to assess the local findings intraoperatively and still isolate and biopsy the possibly displaced soft tissue sarcoma. In conclusion and with focus on a simple technique, a high diagnostic accuracy while having less complications the meta-analysis found CNB to be the superior method .In this study, all biopsies were performed in general anesthesia and by means of a surgically incisional biopsy. The incisional biopsies were highly accurate with a sensitivity of 97.6% for malignancy. In the 2.4% cases, in which a reliable histopathological diagnosis could not be made, re-biopsy was performed with evidence of malignancy. Compared to the results in literature, this can be rated as at least equal, if not even superior , Piya 1, 13.Hau et al. studied the diagnostic accuracy of computed tomography (CT)-guided biopsies and fine needle aspirates of musculoskeletal lesions. They figured 359 cases out, the overall accuracy was 71%. The accuracy for 101 fine needle aspirations was 63% and for 258 CT-guided core biopsies was 74% .Other studies also described that IB can have an advantage over CNB, especially in the biopsy of soft tissue tumors. Pohling et al. described in the case of soft tissue tumors a sensitivity of 100% vs. 81.8% in the comparison of CNB vs IB . RegardiSurgery-specific risks are hematoma and surgical site infection and in relation to CNB — more pain. One also needs to consider the need for re-biopsy in the case of incorrect or unevaluable histopathology as a relevant complication and rated it as such in our study. Many authors recommend CNB over IB precisely because of the supposedly lower complication rate as well as the lower costs , 13, 9.p = 0.003) [In all IB performed in this study, 8 complications occurred, of which 2 were secondary bleeding that required another operation and 6 were re-biopsies because there was no histopathological valid result. In the analyzed collectivity at the UCC was a very low risk of complications of 2.4%. The number of necessary re-biopsies with 0.6% is to be highlighted here, since from our point of view these must also be evaluated as a complication. Klein et al. showed in their study that the number of necessary re-biopsies was significantly higher in the case of CNB compared to IB  .The need of re-biopsy must be taken to account, especially in the case of malignant tumors since these delays further therapy. Also, in the Adams et al study, 6% of the diagnostic biopsies yielded no result (= re-biopsy necessary). Even more critical for patient’s welfare was a 3% rate of false diagnosis. In these cases, the biopsy described a benign tumor, which turned out to be a malignant tumor after complete resection. Patient´s survival of cancerous disease related directly to the speed of treatment .Thus, an appointment for biopsy and consecutive discussion of the patient in the multidisciplinary tumor conference must be available in short time. .A biopsy could be carried out just 8,8 days after the first presentation in the UCC and the final histopathological result was available after a further 10.6 days. Accordingly, the diagnosis was already available 19.4 days after the first presentation. The biopsies of this study obtained by IB further corresponded histopathological in 100% with the histopathology of the resected material, correspondingly there was a sensitivity of 100% for malignancy and a specificity of 97.6% for the tumor type. This also shows how accurate and correctly performed IBs are.Although there is not much detailed data for comparison in the current literature, we see this as a respectable time course that enables patients to be referred quickly to the appropriate individual further treatment.Aside from the already described necessity for re-biopsies, there were 2 postoperative bleedings in our collective that required another surgical treatment. Interestingly, these cases were not associated with a significantly longer operation time in comparison: 15 min and 24 min. All biopsies showed an average operation time of 23.5 min. There were no further complications with the general anesthesia in any case. The extended operation time of the complication courses can be best explained by a more invasive surgical procedure because of a more complex location of the tumor.As already described, due to the extremely large heterogeneity of possible bone and soft tissue tumors of the musculoskeletal system, the type of biopsy is possible in different ways and, as already discussed leads to different results especially regarding the precision of a CNB compared to an IB. Accordingly, there are different recommendations in the literature. In the analysis of patient age performed, the validity of the data is evident. On average, the malignant bone tumors are more than 20 years younger than the patients in whom a metastasis of malignant primaries was secured. This statement agrees with the literature due to osteosarcomas and Ewing’s sarcomas occurring most frequently in young adulthood . The freUltimately, an individual indication for each patient must be carefully examined. In the case of easily accessible tumors, the incision biopsy is usually a harmless method to perform with low complication and high accuracy. In the case of deeper-lying masses that may require complex and more invasive access due to their location, with corresponding soft tissue damage to be expected the possibility of a minimal invasive CNB should be examined. In case of increased risk of postoperative bleeding, CNB can be advantageous.The overall aim should be to guarantee the respective patient the fastest and safest possible diagnostic method and thus promptly supply a definitive therapy, after a thorough discussion of the patient in a multidisciplinary tumor board.All limitations of retrospective studies apply to this analysis. A heterogenous group of surgeons performed the IBs under supervision of an experienced musculoskeletal surgeon. The malignant specimen was resected only by a specialist with more than 5 years of experience in the field of musculoskeletal tumor surgery.Not least due to this work, the different biopsy methods are controversially discussed regarding their advantages and disadvantages. We recommend IB due to the described highest sensitivity, while in summary CNB and other more minimally invasive methods certainly have advantages in surgical effort and lower risk of complications.Ultimately, an individual indication must be carefully considered for each patient. In particular, the surgical access route, as well as the patient’s comorbidity, seems to play a role here. For easily accessible tumors, IB is usually a harmless method with low complication and high accuracy. For deeper masses, which may require a complicated and more invasive approach due to their location, with corresponding soft tissue damage expected, the option of minimally invasive CNB should be explored. If there is an increased risk of postoperative bleeding, CNB may be beneficial.In any case, the goal should be to provide the respective patient with the fastest and most reliable diagnosis possible and thus deliver a definitive therapy in a timely manner. For this reason, guidelines for confirming the diagnosis of soft tissue sarcomas, bone sarcomas, lymphomas, and musculoskeletal metastases should continue to include IB as the gold standard.


Systemic scleroderma (SSc) is a chronic autoimmune disorder that can affect various organ systems. About 90% of patients with SSc have gastrointestinal (GI) manifestations, with esophageal dysmotility being the most frequently reported. While esophageal involvement is the most common, other segments of the upper GI tract can be affected as well, such as the stomach or small bowel. Some of the examples of gastric involvement include gastric antral vascular ectasia (GAVE) and gastroparesis. Small bowel involvement can present with reduced contractility, pseudo-obstruction, small intestinal bacterial overgrowth (SIBO), and atrophy. Although many of these manifestations bear little clinical urgency, acute gastric dilation or pseudo-obstruction constitute a medical emergency and require prompt intervention. We are presenting a case of acute gastric dilation with gastric volvulus in the setting of SSc, which is not well reported in the medical literature. We hope to increase providers’ awareness of this rare manifestation of SSc to facilitate prompt diagnosis and intervention. Furthermore, we hope to prompt more research to be done to better understand its pathophysiology and determine whether this manifestation of SSc is preventable. Systemic scleroderma (SSc) is a chronic autoimmune disorder characterized by vascular and fibrotic changes affecting both the skin and internal organs. Approximately 90% of patients with SSc present with gastrointestinal (GI) manifestations, with esophageal dysmotility being the most frequently reported . The mosA 72-year-old female with interstitial lung disease (ILD) and a well-controlled SSc on mycophenolate presented to the clinic with two days of worsening nausea, non-bloody emesis, and severe diffuse abdominal pain and distension. The patient had no prior abdominal surgeries. Prior workup had revealed a dilated esophagus, megaduodenum, and delayed gastric emptying, but symptoms were well controlled on proton pump inhibitors (PPIs) and a low residue diet. On presentation, laboratory results were remarkable for leukocytosis and lactic acidosis. An abdominal computed tomography (CT) revealed a markedly dilated stomach with a possible volvulus Figure .Laparotomy confirmed acute gastric dilation and volvulus, causing necrosis of the greater curvature of the stomach. Gastric volvulus correction with partial gastrectomy was performed with the subsequent placement of a PEG tube for decompression. The patient’s condition stabilized, and the PEG tube was later reversed.Acute gastric dilation complicated by gastric volvulus in patients with SSc is not well documented in the medical literature. Its pathogenesis remains unclear. Based on our understanding of SSc pathophysiology, the likely underlying mechanism of acute gastric dilation in this patient is multifactorial and includes increased collagen deposition and fibrosis, auto-antibodies affecting myenteric neurons and neural cholinergic-mediated contraction, vascular ectasia, and decreased blood flow ,3. The cIt is also possible that acute gastric dysmotility and gastric volvulus in an SSc patient may follow a pathophysiologic mechanism similar to that of a small intestinal pseudo-obstruction. Some of the common precipitants of small intestinal pseudo-obstruction are electrolyte derangements, anticholinergic medications, and elevations in inflammatory cytokines. While our patient was not on any anticholinergic medications and did not have any electrolyte derangements, we wonder if inflammatory cytokines could have contributed to her presentation. Subsequent volvulus likely occurred around the attachment of the stomach to the gastrohepatic, gastrocolic, gastrosplenic, and gastrophrenic ligaments . It is pManagement options for gastric volvulus depend on the degree of gastric obstruction, presence of ischemia, and comorbid conditions of the patient. In the case of an acute presentation of gastric volvulus, initial management includes intravenous fluid resuscitation and nasogastric tube placement for decompression . DefinitAcute gastric dilation and gastric volvuli are medical emergencies and manifestations of SSc that are not well reported in the available medical literature. We hope to increase providers’ awareness of this rare manifestation of SSc to facilitate prompt diagnosis and intervention. We also hope to prompt more research to be done to better understand its pathophysiology and determine whether this manifestation of SSc is preventable.


Cardiac papillary fibroelastoma (PFE) is a rare tumor, and especially rare when found on the pulmonary valve.We report the case of a 70-year-old woman patient with a pulmonary valve PFE diagnosed incidentally during a follow-up of aortic regurgitation. Computed tomography and magnetic resonance imaging showed no suggestive signs of malignant tumors, and thrombus or myxoma was initially suspected. However, an initial transthoracic and transesophageal echocardiogram did not exclude the possibility of a malignant tumor attached to the wall of the pulmonary artery. Considering the embolization risk, we opted to perform tumorectomy, in which additional surgical procedures could then be conducted if intraoperative diagnosis showed a malignant tumor. Indeed, intraoperative findings showed the tumoral mass attached on the left semilunar cusp of the pulmonary valve, and intraoperative diagnosis of the tumor showed no malignancy. Planned tumorectomy was performed concomitantly with AVR. The pathologic examination of the removed tumor confirmed the diagnosis of PFE. Her postoperative course was uneventful without any sign of recurrence.This case highlights the difficulty of accurate diagnostic imaging and provides valuable insight into a successful surgical treatment of pulmonary valve PFE without any complications. Cardiac papillary fibroelastomas (PFEs) are benign tumors of the endocardium, which account for 7–8% of all cardiac tumors . CardiacA 70-year-old female during a follow-up of moderate aortic regurgitation (AR) was transferred to our institution for further investigation of an undiagnosed cardiac tumor around the pulmonary artery. She had a past medical history of hypertension and hyperlipidemia and was a former smoker. Initial physical examination findings showed stable hemodynamic values and a diastolic murmur at the left sternal border. Laboratory values on admission showed all chemical parameters including D-dimer, soluble fibrin monomer complex, interleukin-2, and tumor markers were within the normal range. Electrocardiogram was unremarkable. An initial computed tomographic CT) scan revealed an irregular tumor (17 × 14 × 8 mm) located on the wall of the pulmonary artery (Fig. T scan reThe procedure was performed under general anesthesia. Cardiopulmonary bypass was performed through cannulations of the ascending aorta and right atrium. After incision of the pulmonary artery, a 20 mm round tumor was discovered Fig. A, and ElPrimary cardiac tumors are rare and account for 0.0017–0.33% of all cardiac tumors based on autopsy studies . Myxoma The development of high-resolution imaging, especially TEE, have facilitated rapid diagnosis of PFE in asymptomatic patients. TTE/TEE, CT, and MRI are thought to be effective diagnostic examinations of PFE, especially as the sensitivity and specificity of TTE are reported to be higher than other examinations with measures of 88.9% and 87.8%, respectively . Indeed,Currently, there is limited consensus with regards to the management of right-sided PPE. So far, asymptomatic patients with a non-mobile tumor have been managed conservatively, and then surgical resection was performed to prevent further complications when the patients presented any symptoms , 12. AntThis case provides valuable insight into a successful surgical treatment of pulmonary valve PFE without any complications, highlighting the importance and challenges of accurate diagnostic imaging.


Technical achievements and surgical techniques improvement contribute to the expansion of the endoscopic spine surgery possibilities. However, today there are few reports about the use of percutaneous endoscopy in spinal tumor surgery. A case of percutaneous transforaminal endoscopic removal of the lumbar spinal nerve tumor with intraoperative neuromonitoring is presented.A 59-year-old female was complaining of a left shin and foot pain, weakness, and paresthesia. Preoperative magnetic resonance imaging (MRI) revealed a tumor (neurinoma) at the left L5-S1 intervertebral foramen. Transforaminal endoscopic removal of an extramedullary tumor from an 8-mm skin incision with intraoperative neuromonitoring was performed. Postoperative MRI revealed the signs of total resection of the tumor.The presented case confirms that percutaneous endoscopic removal of lumbar spine intraforaminal neurinomas can be safe and effective. Percutaneous full-endoscopic spine surgery is known for over 30 years . HoweverMain indication for percutaneous endoscopic intervention on the spine is degenerative–dystrophic pathologies . Apart fIn 2012, first reports were published on percutaneous endoscopy for extradural neoplasms of the spine , 14, andA 59-year-old woman admitted to our clinic, with constant left leg pain lasting for 2 years. Over the past 6 months, there was a gradual increase in pain intensity up to 7–8 Visual analogue scale (VAS) scores. Magnetic resonance imaging (MRI) showed a tumor of the left L5 spinal nerve at the level of the intervertebral foramen .Neurological status: moderate paresis of the left foot extensors (3 points), Lasègue's sign on the left, throbbing pain and paresthesia in the L5 dermatome on the left, and no negative sensitive signs.m. extensor digitorum brevis by 30% compared to the right side. As to mm. peroneus longus, tibialis anterior on the left, no spontaneous activity was detected, motor unit potentials were not changed, and the interference pattern was complete. The ENMG data match a mild axonal preganglionic lesion at the L5 level on the left.Electroneuromyography (ENMG) showed decreased amplitudes of motor responses on the left in abduction of We decided to perform percutaneous full-endoscopic resection of the L5 spinal nerve tumor using a left-sided foraminal approach.The patient received total intravenous anesthesia with propofol and fentanyl. Muscle relaxants were used only for tracheal intubation.mm. extensor digitorum brevis (L5), tibialis anterior (L5), and gastrocnemius (S1) on the left]. Filter setting was made as follows: low-pass filter 30 Hz and high-pass filter 3,000 Hz. We used monopolar continuous cathode rhythmic stimulation with rectangular 4-Hz impulses, stimulus time 0.1 ms, and stimulus intensity ranging from 1.0 to 2.0 mA. Cathode monopolar stimulation was made with a modified elongated probe  through the working channel of the endoscope. A standard needle electrode was placed at the edge of the surgical wound as reference. Monopolar stimulation was made during surgical intervention in order to assess the L5 nerve and its conductivity. Direct nerve stimulation during tumor removal at intensity 1.0 mA evoked motor potentials of the target myotome muscles of the L5 motor root. During the tumor removal no parameters of the recorded motor response significantly varied. Spontaneous electromyography at the tumor removal stage recorded patterns of minimal mechanical impact like single-spike waves in mm. tibialis anterior, extensor digitorum brevis.Intraoperative neurophysiologic monitoring included spontaneous electromyography (free-run EMG) and monopolar direct nerves stimulation . Motor-evoked potentials were recorded with needle electrodes within target muscles located by the anatomical myotomes [On 27 October 2021, surgery was performed with the patient in the prone position. Guided by fluoroscopy, a puncture needle 18G was placed to the intervertebral joint L5-S1 through a point located 10 cm to the left of the midline. A guide pin was inserted along the needle, and the needle was removed. A linear cut 8-mm long was made. A soft tissue retractor was introduced into the wound along the guide pin. A working tube with diameter 7 mm was placed along the retractor, after which the pin and the retractor were removed .TESSYS, Joimax® (Germany) endoscope was inserted into the working tube. Further manipulations were controlled by video endoscopy under continuous irrigation with normal saline solution. The intervertebral joint L5-S1 was‘ visualized. A partial lateral facetectomy was performed with a high-speed burr . An expaAfter electrocoagulation on a small portion of the nerve sheath, an incision was made to see moderately vascularized tissue of the tumor, grayish-red in color, of soft consistency . The tumUpon discharge, the patient had a regress in severe pain of the left leg and Lasègue's sign. Postoperative CT confirmed bone resection in the extent of partial lateral facetectomy .The control contrast-enhanced MRI on the next day and 3 months after the surgery verified total tumor resection, with no accumulation of contrast agent .After 4-month follow-up, paresis of the big toe extensor on the left foot remained, up to 3 scores. Occasionally, the patient feels a slight throbbing pain in the L5 dermatome on the left (2 VAS scores).The report above describes one of lumbosacral neurinoma surgical treatment methods. By Kato classifications (1993), the lesion corresponds to intraforaminal neurinoma type II .dumbbell-shaped neurinomas, up to 4 cm in size, in 12 patients  during percutaneous videoendoscopic resection of L5 neurinoma ensured the safety of surgical procedures, made it possible to remove tumors completely and partially retain anatomical integrity of the affected nerve, which made a positive effect on the functional outcome of treatment.There are no previous reports on application of neurophysiological monitoring with direct monopolar nerve stimulation in percutaneous neuroendoscopic interventions on the spine , 21. Perlimited nature of methods of hemostasis and visualization of sources of bleeding;lack of effective methods for sealing of the dura mater during removal of intradural neoplasms;coaxial method of visualization and manipulation;long time required for a specialist to learn the surgery technique.Obvious obstacles to a widespread use of uniportal percutaneous endoscopic surgery for spine and spinal cord tumors today can be formulated as follows , 22, 23:Intense bleeding greatly worsens the video endoscopic image of the surgery cavity and increases the risk of complications . CurrentThere are different methods of sealing the dura mater in percutaneous endoscopic interventions. Conservative tactics for small defects in the dura mater, combined with hypotensive syndrome therapy, appear to be most effective –27. AmonThe above-listed challenges of the surgery methods can be overcome by using percutaneous unilateral biportal endoscopic technique, which has been widely developing in recent years . In partThe presented case herein shows that uniportal full-endoscopic resection of intraforaminal neurinomas of the lumbar spine with intraoperative neurophysiological monitoring is safe and effective. Further study of potential benefits and effectiveness of percutaneous endoscopic removal of spine and spinal cord tumors must involve a larger number of cases within comparative study.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding authors.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided their written informed consent to participate in this study.MK, SM, OM, and DA contributed to the conception and design of the study, the analysis and interpretation of data, and the work draft. MK and SM designed figures and video. VM and DS offered guidance in study design and revised the article critically for important intellectual content. All authors read and approved the final version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Extramedullary (extraosseous) plasmacytoma (EMP) of the small intestine is a rare plasma cell neoplasm (PCN) that presents with abdominal pain, nausea, vomiting, etc. We describe 2 cases of EMP of the small intestine that presented with ileus.A 78-year-old woman and 68-year-old man visited our clinic with complaints of abdominal discomfort and obstruction. Abdominal computed tomography (CT) revealed a thickened lesion with multiple enlarged mesenteric lymph nodes (LNs) in the ileum and duodenum. The female patient underwent segmental resection in the ileum. The male patient underwent Whipple’s operation in the duodenum.Histopathological examination and immunohistochemical staining of resected specimens from the 2 patients confirmed a PCN. In the surgical specimens of 2 cases, immunoglobulin heavy-chain rearrangement was confirmed by polymerase chain reaction amplification, but no Epstein-Barr virus (EBV)-infected cells were found by EBV-in situ hybridization. Bone marrow aspirate and trephine biopsies were negative for the type of PCN. Bone marrow cytogenetics and fluorescence in situ hybridization revealed no abnormalities. Serum β2-microglobulin levels were within normal limits. Additionally, none of the patients showed an M-spike in serum or urine protein electrophoresis. Therefore, the patients were diagnosed with a solitary EMP of the small intestine. The female patient refused treatment. At follow-up 3 months postoperatively, her disease progressed and she newly developed multiple LNs and nodular lesions in the right pelvic side wall. She was treated with dexamethasone. The male patient experienced back pain 25 days after Whipple’s operation. Spine series magnetic resonance imaging revealed an intermediate signal intensity mass in the posterior epidural space from T8/9 to T10. The mass was removed, and the same histologic features were identified as duodenal masses. He was treated with dexamethasone and radiotherapy.EMPs of the small intestine are easy to overlook because they rarely occur in the small intestine. Although surgery is not required for diagnosis, surgical resection can be a good option for EMPs of the small intestine, instead of local radiation therapy. However, close follow-up is required due to the possibility of relapse or progression to plasma cell myeloma. Herein, we report 2 cases of EMP in the ileum and duodenum in a 78-year-old woman and a 68-year-old man who presented with small bowel obstruction.Extramedullary (extraosseous) plasmacytoma (EMP) is a localized tumor consisting of monoclonal plasma cells that arise in tissues other than the bone. Plasmacytoma presents no clinical features of plasma cell myeloma (PCM) and is classified into 2 types: solitary plasmacytoma of the bone (SPB) and EMP. The incidence of EMP accounts for approximately 1% of plasma cell neoplasms (PCNs) and rarely involves the gastrointestinal tract (GI).This study was approved by the Institutional Review Board of Kyungpook National University Chilgok Hospital (KNUCH), and the requirement for informed consent was waived given the de-identification of all patients’ personal information.A 78-year-old woman visited the emergency room with nausea and vomiting for 10 days on July 31, 2021. She had a history of being diagnosed with peripheral T-cell lymphoma occurring in her left lower neck in 1999 and was completely cured. At the time of admission, abdominal and pelvic computed tomography (CT) showed 7-cm long segmental enhancing wall thickening of the ileum with obstructive ileus of the upstream small bowel loops and multiple enlarged mesenteric lymph nodes (LNs) , whereas free lambda light chains showed 327.86 m/L . The kappa/lambda ratio was 0.09 . Quantitative immunoglobulin (Ig) levels were normal . Additionally, bone marrow aspirate and trephine biopsies were negative. Bone marrow cytogenetics and fluorescence in situ hybridization revealed no abnormalities. Therefore, PCM was excluded and a final diagnosis of EMP of the small intestine was made.Positron emission tomography/CT performed 1 month after surgery showed hypermetabolic LNs in the left paratracheal station and inferior mesenteric area. With progressive disease, chemotherapy was recommended, but the patient refused treatment owing to old age and poor general condition. At follow-up 3 months after surgery, her disease progressed and she newly developed multiple LNs and nodular lesions in the right pelvic side wall. She was treated with dexamethasone and is still alive.A 68-year-old man presented to our hospital with melena and abdominal discomfort in September 2020. He visited another local hospital and was treated for a suspected duodenal ulcer on esophagogastroduodenoscopy. However, despite treatment, his symptoms did not improve, and he was admitted to the gastroenterology department in October 2021. Abdominal and pelvic CT revealed wall thickening with an ulcer in the duodenum; multiple lymphadenopathies were also observed Fig. A. Prelim,2 EMP show normal bone marrow with no evidence of clonal plasma cells in radiographical and morphological assessments. In addition, the diagnosis of EMP should have no clinical features of PCM. Infrequently, EMP can progress to develop PCM, occurring in about 15% of EMP.,4 Two-thirds of patients with EMP are men, and the median age at diagnosis is about 55 years.,5 Most EMPs are localized in the mucous membranes of the upper respiratory tract; more rarely, they can affect the GI tract, skin, bladder, LNs, and central nervous system. However, because of the occurrence of extraosseous sites, the differentiation from lymphomas that exhibit plasma cell differentiation  is necessary. In particular, distinction from marginal zone lymphoma with extensive plasma cell differentiation is difficult in the GI tract, skin, and upper respiratory tract, and it may be impossible by morphological assessment. Among the 2 cases, the female patient had a past history of complete cure of peripheral T-cell lymphoma onset in 1999 after being treated and had been monitored during regular follow-up. Therefore, lymphoma was the first clinically differentially diagnosed despite the uncommon symptoms of bowel obstruction on radiographic imaging. To improve the symptoms of bowel obstruction and confirm the diagnosis, she underwent emergency segmental resection of the small intestine. Although the resected specimens were detected with clonal IGH, CD20, CD79a, and PAX5 were all negative, whereas the plasma cell markers kappa and lambda showed strong and diffuse positive results in the immunohistochemical assessment. Based on the immunohistochemistry results, the diagnosis was considered PCN. The male patient showed similar molecular and immunohistochemical characteristics. As such, the distinction of EMP from lymphomas with extreme plasma cell differentiation is crucial because treatment and prognosis differ among them.,7 For this, immunohistochemistry or immunophenotyping can help make a correct diagnosis. However, in some cases, EMP and lymphomas with marked plasma cell differentiation cannot be distinguished with certainty using immunohistochemistry alone. In such cases, molecular alterations may be helpful, although the molecular features of EMP have not been fully studied. The molecular features of EMP seem to be similar to those of PCM, except in the absence of t translocations and MYC rearrangement.,9 EBV infection is rarely found in EMP, but is detected in 50% to 70% of plasmablastic lymphomas.EMP is a rare hematological malignancy that involves organs outside the bone, representing about 1% of PCNs. Of 61 published EMPs of the small intestine, 20 (32.8%) cases were found in the duodenum, 24 (39.3%) in the jejunum, and 17 (27.9%) in the ileum. Almost all of these cases are described as primary, although secondary EMP is more commonly frequent than primary EMP.  Among 41 EMPs of the jejunum and ileum, 40 cases are confined to these anatomic locations, but EMPs of the duodenum often involve the stomach.,11 Their clinical presentations varied, since symptoms are related to the effect of the mass on the surrounding structures.,12 Possible symptoms are abdominal pain, intestinal obstruction, intussusception, nausea, vomiting, constipation, weight loss, malabsorption, GI bleeding, and obstructive jaundice. Our female patient also had EMP of the ileum, which is a primary type, although she had a past history of lymphoma 22 years ago. Her symptoms and bowel obstruction may also have occurred because of the large mass, measuring 10.0 × 8.0 cm. Unlike our case, Alnimer et al reported a case of multiple EMPs of the ileum in a 51-year-old woman who presented with small bowel obstruction and had a previous history of solitary plasmacytoma of the left acetabulum. They described that their case developed multiple EMPs of the ileum 3 years after the diagnosis of SPB. By contrast, our male patient developed metastasis in the left posterior epidural space of T 8–10 spine due to late diagnosis of EMP. He was surgically diagnosed 1 year after experiencing melena symptoms. He also showed partial obstruction due to the large mass, measuring 10.5 × 7.5 cm. Fortunately, metastasis was observed at only 1 site, and after treatment, the patient is now fine.A comprehensive review of the 61 published cases of EMPs of the small intestine, that was conducted as a literature search of the databases of journals, was reported and patients’ characteristics and clinical manifestations were summarized.,14 Especially, EMP is a highly radiosensitive tumor that can be controlled in 80% to 100% of cases with moderate doses  of radiotherapy. However, the prompt surgical resection of the tumor could be another good alternative therapeutic option for GI EMPs, having the lowest recurrence rate.,15 Furthermore, in the light of our 2 cases, it can be an option for accurate diagnosis. If the tumor can be completely resected, the long-term outcome is equal to that of patients receiving radiotherapy alone.,2 However, if complete surgical resection of the tumor is impossible or doubtful and/or if LNs are involved, combined surgery and radiotherapy is recommended.,15As regards treatment, several retrospective studies have reported that the effective treatment for EMP is local radiotherapy. Regional relapse develops in approximately 25% of patients, and progression to PCM occurs in about 15%.,4 In EMP of the small intestine, only 3 cases were reported to progress to PCM.-19 Because the progression to PCM has a worse prognosis, regular follow-up is necessary using clinical, laboratory, and radiographic images.,20EMP has a better prognosis than PCM and SPB, with about 70% of the patients remaining disease-free over 10 years.In summary, we describe 2 cases of EMP in the ileum and duodenum that presented with obstruction of the small intestine. These cases imply that EMP of the small intestine causes a diagnostic dilemma because of its rare clinical presentation and nonspecific symptoms. In addition, it is difficult to differentiate EMP from lymphoma using imaging studies and histological examination owing to its strong resemblance. Therefore, instead of local radiation therapy, surgical resection can be a good option for EMP of the small intestine for final diagnosis and decent treatment. Although EMP has a better prognosis than PCM and SPB, close follow-up is required due to the possibility of relapse or progression to PCM.Conceptualization: An Na SeoData curation: Ki Bum Park, Hyung Jun Kwon, Dong Won Baek, Seung Hyun Cho, An Na Seo.Formal analysis: Ki Bum Park, An Na Seo.Funding acquisition: Ki Bum ParkInvestigation: Ki Bum Park, An Na Seo.Methodology: Ki Bum Park, Hyung Jun Kwon, Ji Yun Jeong, Dong Won Baek, Seung Hyun Cho, and An Na Seo.Project administration: An Na Seo.Writing – original draft: Ki Bum Park, An Na Seo.Writing – review & editing: Ki Bum Park, Hyung Jun Kwon, Ji Yun Jeong, Dong Won Baek, Seung Hyun Cho, and An Na Seo.


Enterogenous cyst (EC) is a rare congenital lesion generally located in the central nervous system, such as in the cerebral hemispheres, posterior fossa, or spinal canal. They are usually benign lesions, and malignant transformation is rare. A 42-year-old woman felt an obvious pain in the lump and went to a local hospital for local lumpectomy. After 7 months, she again felt pain in the buttocks and difficulty in urinating and defecation. The computed tomography (CT) scan showed a mass in the pelvis. Sacrococcygeal cyst excision was performed 10 days later, and postoperative pathology showed epidermoid cyst. Shortly after, the patient recovered and was discharged from the hospital; the pain in the buttocks continued to recur. Puncture and drainage were performed five times. Later, the patient went to our hospital for treatment, and pelvic MRI showed multiple abnormal signal shadows in the presacral and sacrococcygeal regions, some of which were considered abscesses, and some were cystic lesions. She underwent tumor resection and was diagnosed with EC with locally moderately differentiated adenocarcinoma. Four months later, the patient’s symptoms of swelling and pain recurred. MRI examination showed multiple high-signal T2 shadows in the anterior sacral and subcutaneous tissues of the buttocks, and enhanced scan showed partial marginal enhancement. After assessment, the patient was given a radiation dose of 60 Gy/25F. ECs in the anterior sacral and soft tissue of the buttocks are very rare, and the case of carcinomatous transformation has never been reported. Therefore, we discussed the clinicopathological features of ectopic ECs and reviewed the literature. Enterogenous cyst (EC) is considered as a rare disorder of dermal development. EC occurs during the third week of intrauterine life and evolves from the remnant or ectopic tissue of the neural tube and the gastrum . The tumWe reported a case of EC with adenocarcinoma transformation in presacral and subcutaneous soft tissues and reviewed relevant literature to collect its clinical characteristics, providing reference for the diagnosis and treatment of such patients.A 42-year-old woman revealed that, during childhood, a soft tissue mass near the anus was found. The mass was about 4 cm × 5 cm in size, with a slightly tough texture, and she did not experience pain; thus, she received no special attention. In 2019, she felt an obvious pain in the lump and went to the hospital for local lumpectomy. After surgery, a deep sinus was found, and the doctor repeatedly irrigated the lump with normal saline. After 7 months, she again felt pain in the buttocks and experienced difficulty in urination and defecation. She was readmitted to the hospital, and the computed tomography (CT) scan showed a mass in the pelvis. Vaginal puncture and drainage guided by B ultrasound was performed to relieve difficulty in defecation. Sacrococcygeal cyst excision was performed 10 days later, and postoperative pathology showed epidermoid cyst. The pain in the buttocks continued to recur after the patient was discharged from the hospital. After that, puncture and drainage were performed five times under the guidance of B ultrasound. The last time a drainage tube was placed in the tumor, about 25 ml of yellowish clear liquid was drawn out every day. Later, the patient went to our hospital for treatment.Pelvic magnetic resonance imaging (MRI) showed multiple abnormal signal shadows in the presacral and sacrococcygeal regions, some of which were considered abscesses. MRI scans showed a multiloculated T1 hypointense and T2 hyperintense cystic lesion in the right buttock tissue and sacrococcygeal region. Annular enhancement of the cystic wall was observed on the T2-weighted gadolinium-enhanced MRI scans Figure 1The final histological diagnosis was EC with locally moderately differentiated adenocarcinoma Figure 3Four months later, the patient’s symptoms of swelling and pain recurred. Considering the recurrence of the lesion, the patient was given a radiation dose of 60 Gy/25F. For the rare disease, there is insufficient evidence on the role of adjuvant chemotherapy. Given the immunohistochemical results, a fluorinated pyrimidine- and oxaliplatin-containing regimen was recommended, but the patient declined chemotherapy. We plan to identify KRAS, NRAS, and BRAF mutations and evaluate MSI status to guide subsequent individualized therapeutic and oncological prognosis for the patient, which failed due to the patient’s financial reasons. As of 20 July 2022, the patient had a good outcome, and a recently repeated MRI scan did not show any evidence of cyst recurrence. This indicates that radiotherapy shows a good local control effect.ECs are considered congenital abnormalities. The etiology and pathogenesis of ECs are not clear. Most scholars believe that the ectoderm and endoderm are tight during the early embryonic period. At the third week of the embryo, they separated with the development of the embryo , and the middle was only linked by the nerve-intestinal sac zone. If the embryo development separation disorder, residual or ectopic, it will form perineural EC, often combined with gastrointestinal, spinal, spinal cord and other malformations .ECs mostly occur in the central axis, with cervical segment and upper thoracic segment being more common, lumbosacral department being rare, and other parts being rarer . The cliThe occurrence of ECs in the anterior sacral and hip soft tissue is very rare, and ECs with malignant transformation are rarely reported. We reported a case of sacral caudal EC with carcinogenesis. After a PubMed search, we retrieved a total of nine literatures in The other part included seven cases of ECs undergoing malignant transformation from the central nervous system and retroperitoneum –12, whicin situ lesions may lead to malignant transformation  were all positive, while CK7 was negative, suggesting a foregut origin. CDX2 is a gut-specific nuclear transcription factor and a key regulatory protein for intestinal epithelial formation and differentiation. In gastrointestinal tumors, it is mainly expressed in the small intestine and colorectum, which can be used as a differential indicator of the primary site of tumors. CK20 and the Merkel cell-derived marker of gastrointestinal epithelial transitional epithelium, used in gastrointestinal adenocarcinoma, are often positive for bowel cancer. CK7 (cytokeratin 7), a marker of epithelial origin, is usually expressed in adenocarcinomas, expressed in glandular and transitional epithelial cells, and not expressed in cells of non-epithelial origin. Adenocarcinoma of the gastrointestinal tract was negative. SATB2 is strongly expressed in adult lower gastrointestinal tract and colorectal epithelial cells. SATB2 has a good positive rate in colorectal cancer, and its sensitivity and specificity are high. CK7–/CK20+ carcinomas make up over 90% of cases of colorectal carcinomas  and chemotherapy (including carboplatin and etoposide), but died 1 year after the second surgery . In addiotherapy . In the otherapy , 9–12. FSince ectopic ECs are rarely reported and easily misdiagnosed as other diseases, more cases are needed to collect and summarize their characteristics. Most cases reviewed in this paper are from case reports, and the standards are not uniform, leading to limitations in our conclusions. Further observation and comparative studies are needed.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.HD and DX are the common first authors and the major contributors in writing the manuscript. SZ and ML edited the manuscript and approved the final version. XZ and MF were responsible for reviewing the literature and collecting the information of the patients. All authors listed have made a substantial, direct, and intellectual contribution to the work and approved it for publication.


A lipoma in the mouth is an asymptomatic slowly growing benign tumor of mesenchymal origin consisting of fat.These lesions are soft, and rarely develop in the mouth; in this site, 50% of these tumors may be found in the jugal mucosa or the vestibuleThe etiology of lipomas is uncertain; some authors have suggested endocrine, traumatic, and hereditary causesThe diagnosis is made by pathology of an incisional or excisional specimen. An important feature is that the tumor tends to float when placed in a 10% formaldehyde solutionThe purpose of this study was to report a case of a patient with a large oral lipoma; treatment consisted of surgical excision. The study includes a case report and a review of the literature.J.B.V, a 58-year-old white male patient from the city of Maceió, AL, was referred to the stomatology unit of a dentistry school in the state of Alagoas; he presented a nodule in the right region of the mouth. The patient informed that the tumor had grown within the last six months, and that he was unable to fit a lower dental appliance, which affected chewing and speech; he reported no pain. A large intraoral smooth well-defined sessile nodule of similar color to the surrounding mucosa was observed in the jugal mucosa, a little above the alveolar ridge and the mentonian foramen; it measured about 5 cm on its longer axis. Radiography revealed no bone involvement. An excision biopsy was carried out . The speLipomas of the mouth are benign tumors; they grow slowly, do not infiltrate other tissues, do not ulcerate, and are painless. They are relatively rare in the mouth and the maxilla-facial regionAccording to the literature, mouth lipomas are distributed evenly between sexes; most of these patients are aged over 40 yearsThe diagnosis is clinical and histopathological. The differential diagnosis includes ranulae, epidermoid cysts, pleomorphic adenomas, and fibromas.The treatment, irrespective of histological variation, is surgicalA conservative approach was chosen for the present case; which has so far been effective.It is therefore important to diagnose the lesion correctly in the physical examination and histopathology to establish the prognosis. Healthcare professionals need to understand this disease to treat it adequately.


A mediastinal teratoma is a very rare disease, in which a relatively large mass grows in the space between the two lungs. Although it is a congenital abnormality, it is diagnosed late in the second or third decade. It could be benign or malignant, symptomatic or asymptomatic.A five-year-old child presented with an acute respiratory distress episode due to an acute infection, unresponsive to first-line antibiotics and had a history of similar recurrent pulmonary infections. On imaging, the patient showed an abnormal mediastinal mass which was speculated to be a hydatid cyst or abscess; however, it was later diagnosed during surgery as a mature mediastinal cystic teratoma.Mediastinal tumors are slow-growing tumors that are mostly benign and asymptomatic, but sometimes they may grow, reaching a state where they can affect the neighboring anatomical structures, causing symptoms, which may lead to misdiagnosis.Teratomas, as a differential diagnosis, should be considered in children with recurrent pulmonary infection and radiological findings that resemble masses in the chest. This emphasizes on the importance of performing detailed radiological and lab investigations to set an appropriate diagnosis to ensure a better treatment and prognosis. •Mediastinal mature cystic teratoma in a child•Misdiagnosis is common in mediastinal teratoma, such as: hydatid cyst and lung absence.•Mediastinal teratoma must be considered as a differential diagnosis in mediastinal lesions.•The necessity of doing a CT in multiple recurrent and unresponsive pneumonia These teratomas have an equal distribution in both genders, and mostly get diagnosed at the second and third decades of life 2A five-year-old female child admitted to the hospital due to fever (40 °C), dyspnea and chest pain spreading to the right shoulder and arm. The patient had history of recurrent pneumonia since the age of 6 months, which was always treated using antibiotics. Heart rate, blood pressure and oxygen saturation were within normal range. Clinical examination showed diminished respiratory sounds in the right lung field, mainly at the base, with dullness on percussion with increased resonance.9/L / N=74% / L=21%), platelets count , and hemoglobin was decreased (10.4 mg/dl). Electrolytes were within normal range. Chest X-ray (CXR) showed diffused opacification in the middle and lower right lung field. A computed tomography . Clinical improvement was noticed in the following days with no radiological improvement on the CXR. Therefore, a contrast CT scan was repeated which showed a 50 mm diameter cyst near the heart, with calcified walls; regressed alveolar infiltrates were also noticed in comparison to the previous CT .Fig. 1ThBased on the current findings, the child was transferred to the “Children's University Hospital” so that the mass can be further studied. The child regiment was changed again to (gentamicin and amoxicillin) upon admission. Based on the radiological findings, the diagnosis of Hydatid cyst or lung abscess was considered most likely as they are common in eastern Syria. However, serum antibodies (Echinococcus Ab 1H) were ordered for hydatid cysts which yielded negative results.Nevertheless, a surgical approach was deemed appropriate. The procedure was done through an anterior lateral incision on the fifth intercostal space. A mass was found filling the mediastinum in between the heart and the aorta, with adhesions to the pericardium but no adhesions to the lung. The mass compressed the left lung entrance, with no noticeable effects on the heart.During the procedure, the mass was treated as if it was an abscess, and during the drainage, a clear viscous liquid was extracted from it. The mass was measured (5.5 ∗ 5.5 ∗ 2.5 cm) and connected to another triangular mass measured (3 ∗ 2 ∗ 1 cm), and it was completely removed after releasing the adhesions .Fig. 2ThPathologically, a presence of different sized cystic formation was found, with varying epithelial layers , mucous glands, pancreatic islets, connective tissues with chondrocytes and adipose, cartilage and osseous tissue with granuloma  + 4.Fig.Based on the pathological findings the diagnosis was made as mature cystic teratoma with a secondary infection.Due to low resources, it was decided to do an αFP test only after the surgery, which returned a value within the normal range (1.4 IU/ml), supporting the pathological diagnosis and ensuring that the cystic teratoma is mature and has no malignant aspect.The thorax drainage tube was removed, and the patent was later discharged with a good general condition. A two years' follow-up was done.3Mediastinal teratoma presents in children typically as an anterior mediastinal mass. Other differential diagnoses for an anterior mediastinal mass in children can be thymoma, lymphoma, thymic cyst, lymphangiomas, neurogenic tumors, germ cell tumors and mesenchymal tumors Mediastinal benign teratomas are well-circumscribed heterogeneous masses that have radiographic characters of multilocular cysts with solid and fatty components in addition to calcifications. These radiographic characters may not be present in every case and only 20–40 % of cases show evidence of calcifications Overall, 60 % of patients with mediastinal teratomas are asymptomatic and get diagnosed incidentally during a CXR for other indications Determination of serum tumor markers such as αFP and CXRs are the initial imaging procedure in diagnosing mediastinal teratomas. Moreover, CT scan is the next step in evaluation for specifying multiples factors such as the nature, location and relationship of the tumor to the other structures. Recently, MRIs have shown a superior diagnostic accuracy over CT scans, particularly in identifying tumor invasion to the capsule and to adjacent structures The histopathological examination gives the definitive diagnosis. The differential diagnosis includes hydatid cyst, fungal ball, lung abscess and lung parenchymal tumor Complete surgical excision with delicate separation from the surrounding tissue is the treatment of choice for benign mediastinal teratoma 4We present a child who had recurrent pneumonia during an extended period, which were managed by antibiotics, but later became resistance against first-line antibiotics. Delaying the CXR and the CT scan in such as case caused a delayed diagnosis of a mediastinal mature teratoma.The patient had symptoms and radiological findings of a hydatid cyst, despite having negative serum antibodies; or a lung abscess; both diagnoses were likely due to their etiology and pathology in Syria. However, a teratoma was found to be the cause for the recurrent presentations, which raises the necessity to put benign and malignant tumors in the deferential diagnosis for chest symptoms.WBCsWhite blood cellsNNeutrophilsLLymphocytesPltPlateletsHbHemoglobinCTComputed tomographyAFPAlpha-fetoproteinCRPC-reactive proteinHCGHuman chorionic gonadotropinMRIMagnetic resonance imagingThe case has not been presented at a conference or regional meeting.Patient's parent consent was taken for publishing this case and the images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.Ethical approval was taken from Damascus University's Deanship.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Ayham Ghareeb.This case report is not a first time of reporting, new device or surgical technique. So I would not need a Research Registry Unique identifying number (UIN).AyG: Conceptualization, Methodology, Resources, Data curation, Writing - Original draft.FA: Validation, Writing - Review & editing, Visualization.AA: Conceptualization, Methodology, Software, Writing - Original draft, Formal analysis.MD: Validation, Formal analysis.AmG: Investigation, Software, Review & editing.HD: Supervision, Writing - Review & editing, Project administration.We have no conflict of interest to declare.


Spinal subdural hematoma (SSDH) associated with cranial subdural hematoma (CSDH) is considered extremely rare and the etiology remains unclear. Herein, we report two cases of spontaneous SSDH concomitant with CSDH, with no history of trauma. First, a healthy 35-year-old woman suffered from left leg pain following a headache caused by acute CSDH. Magnetic resonance imaging (MRI) of the lumbar spine showed SSDH extending from the L5 to S2 vertebral levels. The leg symptoms were gradually relieved with conservative treatments within two weeks after onset. The SSDH was completely resolved six months after onset on MRI evaluations. Next, a 69-year-old woman developed a headache and right hemiparesis. Brain computed tomography (CT) demonstrated chronic left-sided CSDH and she underwent a single burr-hole craniotomy. Three weeks after surgery, she experienced difficulty walking because of severe leg pain caused by SSDH extending from the L3 to S1. The clinical symptoms were completely resolved with conservative treatment within one month after onset. At 3 months follow-up, SSDH disappeared on MRI evaluation. Herein, we presented two cases of SSDH associated with CSDH. In both cases, the leg symptoms of SSDH developed following the onset of CSDH. Given that both patients remained active during the interval between CSDH onset and the appearance of SSDH symptoms, the SSDH was likely caused by migration of the CSDH contents to the lumbar spine because of gravity. Spinal subdural hematoma (SSDH) associated with cranial subdural hematoma (CSDH) is considered extremely rare. Furthermore, the etiology and pathogenesis remain unclear. Previous reports have suggested etiologies involving hematoma migration from the cranial to the spinal sites or a coincidence of both CSDH and SSDH ,2. To ouCase 1A healthy 35-year-old woman, with no history of trauma, experienced headaches and saw a local doctor. Initial brain computed tomography (CT) showed no problems. However, seven days later the headaches worsened, and left buttock to posterior thigh pain appeared. Brain CT revealed left acute CSDH and she was referred to our hospital Figure . PhysicaSince left leg pain persisted after admission, magnetic resonance imaging (MRI) of the lumbar spine was performed at seven days after admission. MRI showed a diffuse hematoma-like mass with a high signal on T1-weighted imaging (T1WI) Figure  and an iThree weeks after onset, headache was relieved and CSDH had disappeared on brain CT. At 1 month after onset, her leg pain was relieved. MRI obtained at 1.5 months after onset showed partial resolution of the mass, which was only located in the S1 vertebral level Figures , 3b. At Case 2A 69-year-old woman with a history of hypertension and type 2 diabetes mellitus experienced headaches without any inciting cause. She visited a local doctor one month later. Brain CT demonstrated chronic CSDH bilaterally and she was prescribed Wu-ling-san. One month later, she developed a headache and slight right hemiparesis and was referred to neurosurgery. She had no history of trauma. On admission, brain MRI showed a left-sided chronic CSDH with a midline shift Figure . NeuroloAlthough her right hemiparesis improved postoperatively, she developed left thigh pain. On lumbar MRI at seven days after onset, a hematoma-like mass with a high signal on T1WI Figure  and an iAt three months after onset, MRI showed complete resolution of the mass Figure .Although SSDH itself is a rare condition, SSDH associated with CSDH is extremely rare and its etiology remains unclear. To the best of our knowledge, 43 cases of simultaneous CSDH and SSDH cases have been reported. Of these cases, 22 were post-trauma, three were post-craniotomy, three were after ventriculoperitoneal shunt placement, and the remaining 15 were spontaneous cases .In general, the pathogenesis of SSDH is related to anti-coagulation, blood dyscrasia, vascular malformation, low cerebral spinal fluid pressure, traumatic injuries, lumbar puncture, and spinal anesthesia . In our Two mechanisms of SSDH development associated with CSDH have been reported. The first involves the migration of the hematoma from the cranial to the spinal compartments ,2. The aNeither of our cases had a history of trauma. Nevertheless, leg pain occurred in both cases after their headaches worsened, indicating that there was a time lag between the onset of headache and leg pain. Moreover, both cases were active without any activities or daily living restrictions after headache onset. It is possible that the hematoma grew when the headache worsened and that the SSDH was caused by the migration of the CSDH contents to the lumbar spine because of gravity. Additionally, in both cases, the hematoma was located in the lower lumbar to the sacral vertebrae, which was predominantly on the tail side. Furthermore, in case 1, the SSDH at the L5-S1 level and a subdural hematoma-like lesion at the L1-L4 level had resolved spontaneously in the same period. These findings also support a mechanism involving hematoma migration from the cranial to the spinal compartments because of gravity.Of interest, Kokubo et al. reported that 1.2% of CSDH patients showed concomitant SSDH and had asymptomatic SSDH . Thus, sHerein, we presented two cases of SSDH associated with CSDH. SSDH associated with CSDH is extremely rare and should be suspected in CSDH patients who develop lumbago and neurologic leg symptoms. There is also potential for the hematoma to migrate from the cranial to the spinal space compartments, particularly if the CSDH patients are active. Doctors should be aware of these conditions and check neurological signs cautiously.


Urethral duplication is uncommon with few cases reported in the literature. We report a case in which a patient presented with discharge from proximal part of penis since childhood and recent history of infection. The diagnosis of pre‐pubic sinus was made and complete excision of the sinus tract was done. Intra operative photograph showing complete sinus, a rare entity, which when diagnosed timely and treated will lead to a better quality of life. He alsoAn imaging study suggested that the opening on the dorsal proximal penis did not communicate with the bladder and normal urethra. Urgent USG revealed a collection of loculated abscesses superior to the urinary bladder. MRI scan was done which showed an enhancing thin‐walled multi‐septated loculated, high T2/STIR signal intensity fluid collection in the retropubic space of Retzius with restriction of diffusion, relation, and edema in surrounding fat, most likely an abscess and minimal altered high T2 FS signal intensity in subcutaneous plane dorsal to corpus cavernosa of the penis, likely urethral duplication Distal‐opens on the dorsal or ventral surface of the penis but does not communicate with the urethra or the bladder.Proximal‐opens from the urethral channel and ends blindly in the periurethral tissue.TYPE I—Blind incomplete urethral duplication (accessory urethra).Type II—Complete patent urethral duplication.It is further divided into two types: A (two meatuses) and B (one meatus).Type III—Urethral duplication as a component of partial or complete caudal duplication.Based on the classification given above, our case falls within TYPE I‐A category. Presentations depend upon the type of anatomic variability of the anomaly. Patients may present with a double stream, urinary incontinence, outflow obstruction, recurrent urinary tract infection, sometimes with sepsis; or there may not be any symptoms.4Duplication of the urethra is often overlooked due to its rarity. Therefore, the treating medical professional need to consider the possibility of having this condition, especially if the patient is to present with the aforementioned signs and symptoms indicative of urethral duplication. This can lead to early and accurate diagnosis, hence resulting in the timely management and better prognosis of the disease. Moreover, this case, with the help of histopathological examination, suggests that prepubic sinus is a variant of dorsal urethral duplication.Sajina Thapa, Shila Awal: information of the case, wrote the manuscript, reviewed the literature, and revised the manuscript for important intellectual content. Sunil Basukala, Narayan Thapa: revised and edited the manuscript. Bikash Bikram Thapa, Sajina Thapa: responsible for the integrity of the work as a whole.The authors declare no conflict of interest.This study did not involve experiments on humans and animals.Informed and written consent was taken from the patient prior to his inclusion in this study.


Cavernous haemangiomas, also known as cavernoma or cavernous angiomas, are clusters of vasculature malformations arising from the endothelial layer of blood vessels. They are commonly found in the central nervous systems, skin, or liver. Rarely, they can also affect adrenal glands, a phenomenon with only 66 cases since the first case was reported in the literature in 1955 and 2018. Adrenal cavernous haemangiomas are typically non-functioning and found incidentally on radiological imaging.Here, we present the case of a 79-year-old male who was referred by a district general hospital to our tertiary centre with an incidentaloma of the left adrenal gland which was first noted in 2014 measuring 6 cm. A repeat computed tomography in 2020 revealed the mass was 20.8 cm. In 2020, the growing mass was causing anaemia and abdominal discomfort due to displacement of the surrounding viscera.The initial radiological impression performed in another hospital of the indeterminate mass was highly suspicious of primary adrenal malignancy. The decision was made to operate prior to biopsy as biopsy was deemed high risk for dissemination of primary malignancy of the adrenal.The patient subsequently underwent a radical open left adrenalectomy in September 2020. The diagnosis of adrenal cavernous haemangioma was made on histopathological examination post-surgery. Published literature on this rare adrenal tumour between 2019 and 2021 is also reviewed in this paper. Incidental adrenal masses, or incidentalomas, are asymptomatic adrenal tumours detected on radiological imaging as part of patients’ workup for other complaints unrelated to the described lesion . The preCavernous haemangiomas are clusters of vasculature malformations arising from the endothelial layer of blood vessels . Very raWe report the case of a 79-year-old male with a left adrenal mass diagnosed incidentally in 2014 in the patient’s local hospital. The patient’s medical history included atrial fibrillation and longstanding multiple pulmonary nodules. He did not have any previous surgery. The index computed tomography (CT) scan in 2014 showed a left adrenal mass measuring 5.5 × 4.8 × 5.6 cm, containing foci of fat, necrosis, and calcification with a portal venous phase of 30 HU Figures , 1b.The patient was counselled but declined further diagnostic and therapeutic intervention as he was asymptomatic. The patient continued to be under annual surveillance and interval imaging in his local hospital. In 2017, imaging revealed gradual progression in the size of the adrenal tumour up to 8 cm. The patient was aware that malignancy could not be excluded at least until a biopsy was done. However, he continued to decline investigation and intervention.In March 2020, the patient again presented to his general practitioner with symptoms of intermittent pain in the left iliac fossa and anaemia, whereby his haemoglobin level was 70 g/L. There were no associated symptoms of bleeding, headaches, palpitations, and changes to his appetite or weight. On examination, there was a non-tender palpable mass in the left iliac fossa.A repeat CT scan in July 2020 Figures -2d reveaThe preliminary biochemical tests prior to surgery suggested a non-functioning tumour. Taking into account the global clinical picture, the adrenal mass was more suspicious of a non-functional primary adrenal cancer and unlikely to represent metastases. The decision to operate prior to biopsy was due to the high risk of dissemination of malignancy in case it was a primary adrenal cancer.Following the patient’s referral to our tertiary hospital, surgical management was recommended following multidisciplinary discussion with the aims of achieving a diagnosis, relieving the patient’s symptoms, and preventing complications such as spontaneous haemorrhage. This was also in line with the recommendation of the European Society of Endocrinology Clinical Practice Guideline on the management of adrenal incidentalomas .The patient underwent an open radical left adrenalectomy where the en-bloc of the tumour was resected through a mid-line incision. The operative time was approximately six hours and 50 minutes, while intra-operative blood loss was approximately 400 mL. The patient had two units of packed red blood cells transfused prior to the procedure and was further transfused two units of packed red blood cells on day three post-operatively. Intra-operatively, the left adrenal tumour was found attached to the spleen, left kidney, and distal pancreas despite there being no invasion of those structures on the most recent radiological imaging. The specimen was subsequently removed en-bloc Figure .Chyle leak was diagnosed on day seven post-operatively from fluid in the patient’s left abdominal drain bag. The patient was managed conservatively with the use of somatostatin analogue (octreotide 100 µg three times a day) together with the dietitian’s modified low-fat diet. Apart from that, the patient’s recovery was uneventful and he was discharged home after 23 days of hospital admission. The chyle leak had resolved when the patient returned for his follow-up drain removal on day 27.On gross pathological examination of the specimen, the encapsulated adrenal mass consisted of the adrenal with attached spleen, left kidney, and distal pancreas. The combined weight of the specimen was 4,060 g. The spleen and left kidney were both attached to the tumour. The encapsulated adrenal mass measured 18 × 13 × 7 cm.On slicing, most of the tumour appeared to consist of necrotic tissue around the edges, of which there was a dark haemorrhagic appearance Figure , Panel bThe encapsulated tumour consisted largely of dilated blood vessels, many of which were filled with recent and organising thrombi. There were large areas of fibrosis and sclerosis with evidence of old haemorrhage Figure , Panel dIn many areas, there was evidence of papillary endothelial hyperplasia of the cells lining many of the dilated blood vessels. The adrenal gland was identified outside the capsule of the lesion, and there was some evidence of atrophy as a result of compression by the tumour. The attached kidney, spleen, and distal tail of the pancreas showed no histological lesion. There was no evidence of malignancy.Adrenal cavernous haemangiomas are rare, non-functioning, benign tumours which are commonly diagnosed with histological evidence following surgery, as seen in our patient. The first case of adrenal haemangioma in a live patient was reported by Johnson and Jeppesen  in 1955.Between 1955 and 2018, only 66 cases have been reported in the literature, reflecting the rarity of this condition. In addition to the literature review by Degheili et al. , seven pOver half of these tumours were initially detected incidentally on imaging as incidentalomas . The majThe summary of cases in Table Overall, 70% of cases had normal metabolic workup, and the same was seen in our case. The open surgical approach appeared to be the choice of management in 71% of cases. Lesions were resected by laparoscopic approach in 18 (24.6%) cases, and laparoscopy seemed to be the preferred option for smaller lesions measuring between 3 cm and 12 cm. Having said that, the largest lesion resected laparoscopically was 27 × 17 × 5.5 cm weighing 2 kg .To our knowledge, there is only one other case reported in the literature with anaemia as one of the symptoms of adrenal cavernous haemangioma . Hence, Reported radiological signs associated with adrenal haemangiomas are heterogeneous internal structures, speckled calcifications , and denHistologically, adrenal haemangiomas are often described as cavernous, well-encapsulated and localised to the adrenal cortex. They have multiple peripheral vasculature dilatations associated with complex necrotic, calcified, fibrotic, thrombotic and haemorrhagic vascular changes . There aMost reported cases of adrenal cavernous haemangioma were treated surgically, and, indeed, the diagnosis was made frequently following histology. As expected, there are currently no definitive treatment and management guidelines on adrenal cavernous haemangioma. However, The European Society of Endocrinology’s guideline on the management of unilateral adrenal incidentaloma recommends adrenalectomy if there is radiological suspicion of malignancy with or without local structural invasion and clinically relevant hormonal excess . ResectiIn our case, the patient underwent a major surgical procedure for a benign tumour. This was because of the initial indeterminate mass on radiological imaging and a high risk of biopsy. It was justifiable that the large tumour was resected as it was causing discomfort to the patient and anaemia.In summary, adrenal cavernous haemangioma is an extremely rare entity. It typically presents as incidentalomas on imaging. The diagnosis of adrenal cavernous haemangioma is done exclusively by histology. Therefore, diagnostic resection of an adrenal mass as clinically indicated is recommended for lesions larger than 4 cm. Resection may additionally relieve compressive symptoms and avoid potential spontaneous haemorrhage.


Neurofibromatosis type 1 (NF1) is a congenital condition characterized by "café au lait" spots and subcutaneous fibromas. There are various combined diseases, such as malignant tumors in the abdominal organs or brain tumors.Here, we present a case of a 35-year-old patient with a rare combination of NF1 with a gastrointestinal stromal tumor (GIST) and pancreaticobiliary maljunction (PBM). At the first visit, her main symptom was right upper abdominal pain. Radiological investigations revealed a common bile duct stone, submucosal tumor in the duodenum, PBM, and abnormal findings in the intrahepatic bile ducts. After the common bile duct stone was removed by endoscopic intervention, the patient underwent laparoscopic cholecystectomy, resection of the duodenal submucosal tumor, and liver biopsy. Pathological examination revealed chronic cholecystitis, GIST of the duodenum, and chronic inflammation of the intrahepatic bile ducts.This is the first case report of the rare coexistence of GIST and PBM in a patient with NF1. Neurofibromatosis type 1 (NF1) is a rare hereditary disease caused by alterations in the NF1 gene .Gastrointestinal stromal tumors (GISTs) are uncommon neoplasms derived from Cajal’s interstitial cells in the gastrointestinal tract . The incPancreaticobiliary maljunction (PBM) is a rare congenital anomaly, defined as the union of the pancreatic and biliary ducts located outside the duodenal wall . AlthougIn this report, we present a rare case of the coexistence of duodenal GIST and PBM in a patient with NF1.A 35-year-old woman with NF1 was referred to the emergency care department of our hospital with intermittent severe epigastric pain. Abdominal contrast-enhanced computed tomography revealed a stone in the lower common bile duct and a tumor in the duodenal bulb Figure . EndoscoBased on these results, laparoscopic cholecystectomy, partial duodenal resection, and liver biopsy were performed. A tumor on the anterior wall of the duodenal bulb was identified during the surgery Figure . We placNF1, historically known as von Recklinghausen’s disease, is an autosomal dominant hereditary syndrome usually diagnosed in childhood or adolescence . NF1 is The most common tumor affecting the gastrointestinal tract in patients with NF1 is GIST ; howeverPBM is an abnormal junction of the pancreaticobiliary ducts that leads to the reflux of bile and pancreatic juice. Because of this reflux, it is known to cause cancers of the bile duct or gallbladder . PBM witIn the present case, MRCP showed irregular shapes and walls of the intrahepatic bile ducts; therefore, a liver biopsy was planned to clarify the underlying disease. There were several possible diseases, such as PSC, IgG4-related hepatitis, and secondary changes due to PBM. The patient’s history and blood test and liver biopsy results did not confirm the diagnosis. Certainly, the bile duct appearance on MRCP suggested the possibility of PSC, but the elevation of serum alkaline phosphatase level was transient, and there were no characteristic findings of PSC in the liver biopsy specimen. According to the diagnostic criteria of PSC, this patient was diagnosed with probable PSC . Either The coexistence of NF1 and PBM is rare. NF1 and PBM are both congenital diseases, but the hereditary cause of PBM is unclear. To the best of our knowledge, only one previous report has shown a combination of neuroendocrine tumor (NET) and PBM with biliary duct dilatation . There aWe present a rare case of the coexistence of duodenal GIST and PBM in a patient with NF1. We were able to provide proper treatment through preoperative examination and consideration of the patient’s background. When seeing an NF1 patient, we should always consider the possibility of a hidden tumor, as well as any abnormal structures in the bile duct. Since the relevance of NF1 and PBM is unclear, further research would be warranted.


Objective: To present an atypical case of tuberculous dacryocystitis. Method: An adult female presented with long standing epiphora with gradual swelling over lacrimal sac region. On syringing, water was felt in throat with no regurgitation. CT-DCG and CECT orbit were done subsequently and simultaneously. Ill-defined, enhancing soft tissue surrounding and involving the lacrimal sac wall was identified. The sac wall outline was seen distorted with contrast in NLD. The histopathology was suggestive of non-specific chronic inflammation. GeneXpert analysis was shown to be very low positive for M. tuberculosis. Montoux test was strongly positive (40 x 40 mm). ATT was started. Results: The swelling and watering subsided over the next few months. Conclusion: Tuberculosis should be considered in cases of chronic granulomatous dacryocystitis. CECT with CT-DCG is essential imaging. GeneXpert is a new and sensitive tool with considerable specificity in cases in which histopathology is not conclusive. ATT is curative and DCR is reserved for only unresolved NLDO with persistent epiphora.  Diagnosing tuberculosis of lacrimal sac can be cumbersome in patients with epiphora. A high degree of clinical suspicion should be kept for patients from endemic regions. Herein, we present a case of epiphora, in which the detailed clinical workup revealed the sac tuberculosis and saved the patient from unwanted (dacryocystorhinostomy) surgery.Fig. 1 A. No redness, warmth, discharge or regurgitation of sac on pressure was noted. Both puncta appeared normal on slit lamp examination. On syringing, water was felt in the throat with no regurgitation or delay. No regional lymph node enlargement, history of fever, immunosuppression or systemic illness were noted.A female in her 40’s came to our outpatient department for a second opinion on her left side dacryocystorhinostomy (DCR) surgery that was advised elsewhere. She presented with left eye epiphora that she had for the last six months. This was associated with painless, gradual swelling over the left medial canthus region. On inspection it appeared as a fullness in the lacrimal sac area, which was firm and non-tender on palpation . CT-DCG and CECT orbit were done subsequently and simultaneously. On CECT, ill-defined, enhancing soft tissue surrounding and involving left canaliculi and the lacrimal sac wall, was identified . The sac wall outline was seen distorted with similar soft tissue enhancement seen inside the nasolacrimal duct (NLD). Minimal post-septal extension  was noted. There was no frank necrotic area or bony change. Paranasal Sinuses appeared normal. On CT-DCG, contrast was seen inside the left sac, NLD and inferior meatus .X-rays Dacryocystography (DCG) revealed patent but mildly irregular sac lumen  incision. The biopsy specimen was taken from the sac wall in full thickness. Sero-mucoid discharge samples from the inside of the sac were collected. Hematoxylin-eosin (H&E) stain, Periodic acid Schiff (PAS) stain and Cartridge-based nucleic acid amplification test  were performed on the biopsy specimen. Gram stain, KOH mount, Zeil-Neilson (Z-N) Stain, Bacterial culture (Blood agar and Chocolate agar) and Fungal Culture (SDA) were performed on both the biopsy specimen and the discharge samples.M. tuberculosis . No resistance to Rifampicin was found. Montoux test was strongly positive (40 x 40 mm induration with redness at 72 hours). Stains and cultures results were inconclusive. Pulmonary physician and Otolaryngologist consultations were done to rule out any other/ systemic foci. Category I Antitubercular treatment (ATT) i.e. 2 HRZE + 4 HR was started for six months. Improvement was noticed in the first month with complete resolution of all symptoms within the first four months . Visual acuity, colour vision, contrast sensitivity and fundoscopy were normal on monthly follow-ups. No investigation was performed at completion of ATT course. No recurrence was reported after one-year drug-free period on three monthly follow-ups.The histopathology was suggestive of non-specific chronic inflammation. Gene Xpert analysis was shown to be very low positive for 1]. This could be because of a relatively high prevalence of primary acquired nasolacrimal duct obstruction (PANDO), non-specific long-standing symptoms, sub-clinical infection, and unproper evaluation with adequate pre-operative clinical or radiological studies especially in failed DCR cases . In our case, the absence of redness, warmth, pain and tenderness ruled out acute inflammation. The firm nature in the absence of discharge or regurgitation ruled out the possibility of dacryocystitis or mucocele. As a standard practice, syringing must be done to rule out mechanical blockade before taking the decision for surgery, which was not mentioned in some previous reported new cases .Sac tuberculosis has been underreported in India, where tuberculosis is endemic. Most of the reported cases were suspected by surgeons while performing dacryocystorhinostomy and were diagnosed post-operatively [3]. X-rays DCG, less informative than CT-scan, can be done , for advantages like less radiation, being cheap and fast, or when the CT facility is not available.CECT and CT-DCG in same sitting help in the further planning in the atypical presentation of epiphora .Microbiology and pathology may not be conclusive in tuberculous lesion, which could be due to low bacterial load, fastidious nature of the bacteria, inadequate sample, etc. GeneXpert (CBNAAT), a real time polymerase chain reaction (PCR), is a rapidly feasible test, highly sensitive and specific in detecting traces of 6].Strongly positive Montoux test (> 15 mm) alone, without identifiable risk factors, is suggestive of active TB. Sometimes, lupus vulgaris overlying sac lesion could raise suspicion of active tuberculosis of the sac and may have a strongly positive Montoux test [4]. Nasal mucosa involvement has been reported in several published cases of sac tuberculosis, many among them being documented after primary DCR surgery [2]. So, whether it was “primary” nasal mucosal lesion or “secondary” lesion spread from sac directly, is not clear yet.It is not necessary to have systemic foci, isolated or primary sac tuberculosis (TB) has been reported, but should be ruled out by pulmonary physician and otolaryngologist . If any suspicious tissue suggesting granuloma, caseous material, bone erosion or mass are found, full-thickness incisional biopsy from the sac wall must be sent for histopathology and microbiology, without proceeding to osteotomy . If the laboratory reports are favorable, then only one can safely proceed to DCR. We believe that osteotomy breaches compartmentalization of the lesion and mucosal anastomosis provides free passage for direct spread of tuberculous granuloma to nasal cavity. As for standard teaching, opening the sac before making the bony ostium, inspecting its contents and inner walls/ sac-flap in full length under operating microscope during external DCR, is essential .Anti-tubercular regime is curative and should be followed as per WHO guidelines. Surgery, either dacryocystorhinostomy or dacryocystectomy, is not indicated primarily and should be reserved for persistent epiphora after completion of ATT course, especially for the epicenter in nasolacrimal duct or inferior turbinate [To conclude, tuberculosis should be considered in cases of chronic granulomatous dacryocystitis. CECT with CT-DCG is essential imaging. GeneXpert is a new and sensitive tool with considerable specificity in such cases in which histopathology is not conclusive.Conflict of Interest statementThe authors state no conflict of interest.Informed Consent and Human and Animal Rights statementAn informed consent was taken from the patient for publishing the clinical photographs.Authorization for the use of human subjectsEthical approval: The research related to human use complies with all the relevant national regulations, institutional policies, is in accordance with the tenets of the Helsinki Declaration, and has been approved by the review board of Oculoplastic and Ocular Oncology Services, IRIS Superspeciality Eye Hospital, Ranchi, India.AcknowledgementsNone.Sources of FundingNone.DisclosuresNone.


Cochlear implant (CI) is a surgically implanted device designed for treatment of severe sensorineural hearing loss in pediatric and adult patients. With the advanced technology and satisfying results, the indications of CI are gradually expanding.A four-year-old male patient with cochlear implant was referred to our clinic with the complaints of pain having occurred during MRI and failure to be able to replace the external part of cochlear implant over the internal part. The 3 T MRI had been performed the day before. According to information received from his parents, the patient had bilateral profound congenital hearing loss. He had undergone tumor resection from his right ear 15 months earlier. Total hearing loss had occurred in his right ear after tumor surgery. He had been diagnosed with Langerhans cell histiocytosis. Six months after the tumor surgery, a cochlear implant (Nucleus Freedom Straight CI24RE) was implanted in his left ear for sensorineural hearing loss. During his follow-up, complaints of excessive fluid intake and frequent urination emerged. Further evaluation confirmed a diagnosis of diabetes insipidus and an MRI was planned with suspicion of intracranial spread of Langerhans cell histiocytosis. A head bandage was applied before the MRI that took place in another center but the patient experienced pain during the MRI procedure; the imaging process was terminated immediately. As the external piece of device could not be replaced, the patient was referred to our clinic.The physical examination showed a swelling in the area where the magnet was estimated to be located. The magnet had turned upside down, the external part was reversed (inside facing out) and still attracting to the internal part . After rA skin incision was performed under sedoanalgesia as it would not pass through the skin contact area of the device's external and internal part . After iThe risk of a complication is very low for cochlear implants. Vestibular complaints 3.9%), device failure (3.4%), loss of taste 2.8%) and skin problems 1.3%) are the most common documented long-term complications in the meta-analysis studies..3% are t.8% and s.9%, deviIn a study published in 2014, Hassepass et al. reported that they had performed 22 magnet revision surgeries out of 2027 cochlear implant patients. Twelve (52.2%) of these patients had a dislocation that had occurred after MRI.The problem faced with the change of a magnet's polarization was solved by changing the direction of external magnet in a case presented by Jeon et al.One of the issues related to cochlear implant users is complications that may occur during MRI, which is widely used as a standard imaging method nowadays. To the best of our knowledge, this is the first case of magnet dislocation seen after 3 T MRI. For prevention of such complications during MRI, patients and their relatives should be informed in detail about the possible risks when using higher resolution MRI.The authors declare no conflicts of interest.