External root resorption is a lytic process occurring in the cementum and/or dentine of the roots of teeth. Although till date the predisposing factor is yet to be discovered, the suspected causes are considered to be peri radicular inflammation due to trauma, granuloma, central jaw tumors, excessive forces, impaction of teeth, and systemic diseases. A 23-year-old male reported to the department of conservative dentistry and endodontics with a chief complaint of pain and food lodgement in lower left mandibular region since 5-6 months. On clinical examination, grossly decayed tooth (#36) which was tender on vertical percussion and was not associated with any mobility. Intraoral periapical radiographic revealed external root resorption in distal root, which was concomitant with mild bone loss. Taking into account the extent and the severity of the resorption the treatment course was planned that included Root Canal Treatment. The distal and two mesial canals were obturated using mineral trioxide aggregate and gutta-percha respectively. After completion of obturation, it was followed by post endodontic composite restoration. The patient was followed-up after 1 year and the tooth was functional without sensitivity to percussion or palpation. Tooth showed normal physiologic mobility and no periodontal pockets on probing. Radiographic findings revealed no periapical changes and no further progression of External root resorption.


Inflammatory myofibroblastic tumors (IMTs) are a group of soft tissue neoplasms with a predilection for the lungs and abdominopelvic cavity, characterized by a mixture of fasciitis-like, compact spindle cells, hypocellular fibrous histologic patterns, and distinctive molecular features. Due to their unspecified symptoms and non-specific radiologic presentation, the histopathologic and immunohistochemical analysis of a biopsy specimen is crucial for the diagnosis. We present a case of a 30-year-old man with intermittent hemoptysis diagnosed with a pulmonary IMT. We aim to review the literature regarding its definition, clinical findings, diagnosis, treatment, and prognosis. The treatment for an IMT is based on its location and extent, including complete surgical resection, which has a good prognosis compared to corticosteroids, chemotherapy, radiotherapy, and non-steroidal immunomodulation in patients who are not good surgical candidates. Further investigative studies with larger sample sizes and longer meticulous follow-ups are needed to demonstrate this neoplastic disease's natural history and find appropriate management for it. Inflammatory myofibroblastic tumors (IMTs) were first described by Brunn in 1937. They represent an extremely rare type of inflammatory pseudotumor that appears most commonly in children and young individuals, with a global prevalence ranging from 0.04% to 0.7%, irrespective of gender or race . AlthougHere, we discuss the case of a 30-year-old male who reported hemoptysis and was diagnosed with a pulmonary IMT. The aim of this report is to present a case of an inflammatory myofibroblastic tumor diagnosed by immunochemistry and review the literature on its definition, clinical findings, diagnosis, treatment, and prognosis.A 30-year-old man with a history of nephrolithiasis presented to the emergency room with complaints of flank pain and an episode of small-volume hemoptysis (two to three teaspoonfuls of blood a day) before presentation. He denied fever, chills, chest pain, night sweats, or weight loss. The patient reported intermittently experiencing episodes of non-massive hemoptysis for the last six years. There was no known history of tuberculosis or valley fever exposure, and he did not have a history of recent travel.On admission, computed tomography angiography (CTA) of the chest revealed a lesion initially interpreted by radiologists as a cavity. However, it was concluded that the lesion was a solid nodule containing rounded soft tissue mass, with a dilated airway mimicking a cavitary change. It was seen to be arising adjacent to the right posterior basal segmental bronchus along with adjacent ground-glass airspace opacities, likely reflecting pulmonary hemorrhage Figure .This lesion appeared to have progressed since a prior CT scan was done five months earlier. Differential diagnoses of the lesions included fungal infection, bacterial superinfection of a preexisting right lung cystic lesion, or a neoplasm such as bronchial carcinoid. Bronchoscopy and bronchoalveolar lavage (BAL) of the posterior basal segment of the right lower lobe (RLL) were notable for some fresh blood emanating from the posterior basal segment of the RLL but no endobronchial lesion. A biopsy was not done. BAL cultures were negative for bacteria, fungus, or acid-fast bacilli (AFB).The patient was subsequently discharged home with plans to follow up on an outpatient basis. An Ion protocol CT chest scan was done three weeks later, which showed a right infrahilar mass measuring 2.4 x 1.7 x 2.4 cm in size, showing an increase in size. Three weeks later, a robot-assisted navigational bronchoscopy with radial endobronchial ultrasound (r-EBUS) and fluoroscopy-assisted transbronchial needle aspiration and brushing of the RLL lung nodule were performed  and anaplastic lymphoma kinase (ALK) Figures , 6.The spindle cells did not express S-100, SOX-10, cytokeratin AE1/3, CAM 5.2, CD56, synaptophysin, chromogranin, TTF-1, desmin, and HHV-8. The morphology and immunoprofile together support the diagnosis of an inflammatory myofibroblastic tumor. The patient was referred to an oncologist as well as a thoracic surgeon for further management, and was lost to follow-up.An inflammatory myofibroblastic tumor is a unique tumor with predominantly benign potential, with a few reports of distant metastasis . It variIMTs are also difficult to diagnose by imaging, as was demonstrated in this case. The rarity of this tumor, along with its non-preference for a specific bodily location, makes the process of diagnosis arduous. However, IMTs grow and can start invading adjacent organs. Hence, the need for an accurate and speedy diagnosis is of the greatest importance. The rate of growth, as well as the nature of the tumor, can be determined by its associated biological markers . It was When involving the lungs, the tumor presents with non-specific respiratory and systemic symptoms, and therefore chest imaging is crucial in identifying lung lesions early. Patients with a similar presentation require a biopsy of the lung lesions either via CT-guided biopsy or bronchoscopy with endobronchial ultrasound and fine needle aspiration for a histological diagnosis. As discussed above, histological examination alone is inconclusive. To differentiate it from various benign and malignant tumors conclusively, immunohistochemistry for ALK expression, fluorescence in situ hybridization (FISH), and next-generation sequencing (NGS) are assistive .The treatment for IMT depends mainly on the site and extent of the tumor. Complete surgical resection is associated with a better long-term prognosis. However, lobectomy and pneumonectomy are preferred over wedge resection or endobronchial resection, as these decrease the risk of local recurrence and metastasis more effectively . A good Chemotherapy, radiotherapy, and non-steroidal immunomodulation have been attempted in patients who were not good candidates for surgery. However, these treatment methods were found to be ineffective against this aggressive tumor . Table 1Inflammatory myofibroblastic tumors are an uncommon subgroup of benign soft tissue neoplasms for which an exhaustive clinical exam, as well as investigative procedures, must be conducted first. In short, inflammatory myofibroblastic tumors may be considered per exclusion. Competitive inhibitors of ALK tyrosine kinase, such as crizotinib, may be used with a variable response if ALK-1 markers are positive. Because of the rarity and non-specific presentation of the pulmonary inflammatory myofibroblastic tumor, as seen in our case, we strongly suggest including the disease as a differential diagnosis when patients present with unclassifiable respiratory signs and symptoms. Histopathological evaluation is necessary for diagnosis and should be done early, given the aggressive nature of the tumor. For higher diagnostic rates and favorable outcomes, further investigative studies with larger sample sizes and longer follow-ups are needed to establish the natural history of this neoplastic disease and discover appropriate medical management.


Acute abdomen caused by spontaneous rupture of the inflammatory myofibroblastic tumor of the urinary bladder (IMTUB) is a rare clinical emergency of the urinary system.It is difficult to distinguish it from spontaneous rupture of bladder caused by other causes before surgery. An emergency case of spontaneous rupture of IMTUB was reported. A 57-year-old middle-aged woman was admitted to the hospital because of “acute lower abdominal pain for 5 hours”. No history of smoking and gross hematuria. The physical examination revealed visible abdominal tenderness as well as signs of shock. A pelvic CT scan shows a space-occupying lesion above the bladder with massive accumulation of blood. When the nature of the tumor could not be determined, emergency laparotomy and partial cystectomy were performed, and postoperative pathology confirmed cystitis myofibroblastic tumor. No local recurrence or distant metastasis of the tumor was observed during the regular follow-up period of 6 months. IMTUB should focus on prevention and treatment, with a combination of preoperative examination and postoperative pathology, and finally implement highly individualized treatment. Inflammatory myofibroblastic tumor (IMT) is a rare clinical benign mesenchymal tumor with uncertain malignant potential that can occur in the lung, gastrointestinal tract, liver, abdominal wall, or retroperitoneal soft tissue, among other places , and lesThe clinical presentation of inflammatory myofibroblastic tumor of the urinary bladder (IMTUB) is characterized by episodic painless hematuria . The patWe present a case of acute abdomen caused by spontaneous rupture of IMTUB. It provides a reference for the emergency diagnosis and treatment of spontaneous rupture and bleeding of IMTUB by reviewing its diagnosis and treatment process, in combination with relevant literature reports.The patient, a 57-year-old female farmer, came to the clinic on August 14, 2021. She developed severe pain in the lower abdomen with no obvious cause 5 hours ago, accompanied by nausea and vomiting. The outpatient was admitted to the hospital because of “abdominal pain, the cause of which is being investigated?” On admission, the blood pressure was 88/56 mmHg, and signs of shock were visible. Physical examination revealed a distended abdomen with obvious tenderness but no rebound tenderness. A pelvic CT scan showed that the bladder can be filled, there is a 7.4cm-diameter spherical kind of oval low-density shadow above the bladder and a space-occupying lesion above the bladder, as well as large number of pelvic lesions with massive accumulation of blood about 1000ml.  is relatively slow, and patients frequently present with intermittent or persistent painless gross hematuria as the first symptom , with loIMTUB is typically seen as a smooth-walled, wide-basal lesion rather than a papillary lesion. Lamovec accurately described the IMTUB histological pattern. The tumor’s luminal surface was edematous and focal hemorrhagic fibroblast tissue, with a large number of capillaries and sparsely cellular fibromatous tissue, accompanied by inflammatory cells, plasma cells, and/or lymphocytic infiltration . The anaPathological findings are still the gold standard for determining IMTUB. Vimentin, ALK, and SMA are mostly diffusely positive in IMTUB pathology, but not S100, CD117, CD34, STAT6, and so on . ALK is Ki-67 positivity is an important factor in predicting benign and malignant tumors, recurrence, and progression, and higher Ki-67 expression often indicates a higher malignant tendency of tumors  and is aIn this case, the patient developed hypovolemic shock as a result of spontaneous rupture and bleeding of the IMTUB and required an emergency partial cystectomy. The patient received good surgical benefits, according to postoperative pathology and follow-up results. Currently, surgery will provide the greatest benefit for this condition, and the clinical treatment of IMTUB is highly individualized, and the common surgical methods include transurethral resection of bladder tumor (TURBT), partial cystectomy and radical cystectomy. Some researchers have performed intravesical instillation of anti-inflammatory drugs, but no clinical significance has been established . The WHOThis case demonstrates that, in addition to advocating for individualized treatment, IMTUB treatment is more important to early diagnosis and intervention. Because of the patient’s lack of awareness of physical examination, the patient’s final illness was delayed, and the acute abdomen caused by spontaneous rupture and bleeding of IMTUB posed a serious threat to the patient’s life and made diagnosis and treatment difficult. We recommend that patients with abdominal pain, gross hematuria, abdominal mass, or other unpleasant symptoms seek medical attention and treatment as soon as possible to avoid delaying treatment. The ultimate goal is to accomplish the goal of early detection and treatment.Due to the difficulty of biopsy under the microscope with IMTUB, TURBT can be used for pathological examination to determine the nature of the tumor and choose a surgical plan. When the early examination found that the tumor was small, single, and the first electroresection pathology shows no muscle layer invasion, the bladder tumor electroresection (TURBT) is a viable option. However, due to its relatively high recurrence rate, regular follow-up of cystoscopy and pelvic CT are required every 3 months after the operation. Partial cystectomy is recommended when the tumor is large and the pathology shows that the muscle layer has infiltrated, but the tumor can still be completely removed. Radical cystectomy is an option when the tumor is multiple in the bladder or has occupied most of the bladder and cannot be completely removed, However, the quality of life and psychological problems of patients after total cystectomy must be closely monitored. As a result, partial cystectomy has become the preferred surgical method for treating IMTUB.In terms of clinical symptoms and signs, spontaneous IMTUB rupture is difficult to distinguish from spontaneous bladder rupture. It can be distinguished from clinical symptoms and imaging examination. The first signs of spontaneous bladder rupture include severe abdominal pain, massive hematuria or microscopic hematuria, as well as obvious local tenderness, positive rebound tenderness or no obvious signs. The CT plain scan shows poor bladder filling, and significant contrast agent leakage can seen in the pelvis. The main cause of its occurrence is the pathological changes of the bladder itself, such as tumors and diverticulum . At the IMTUB is a rare disease, and spontaneous rupture requires a combination of preoperative examination, intraoperative findings, and postoperative pathology, as well as multiple differential diagnoses. It should be considered as one of the causes of spontaneous bladder rupture. Before surgery, the spontaneous rupture of IMTUB must be distinguished from other diseases by combining the medical history and auxiliary examination. Among many treatment options, partial cystectomy stands out for its low recurrence rate, quick recovery, and high quality of life. The final surgical method should be determined by imaging analysis, preoperative histopathology, and intraoperative conditions. The pathological results of TURBT are especially important for the selection of surgical methods in non-emergency patients with IMTUB. Early examination, regular review, and individualized treatment are recommended for patients with unexplained abdominal pain and hematuria. To improve the detection rate of IMTUB, future research should focus on the detection of blood and urine markers, as well as more targeted imaging examinations.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.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. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.ZS, LQ and YZ have made substantial contributions conceptions and design and have been involved in drafting the paper. ZG, HG and XH have performed data acquisition. ZS, YD, WY revised the manuscript. All authors contributed to the article and approved the submitted version.The authors thank the family for participating in the study.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.


Due to the conspicuous morphology of the deformity and the fact that primary reconstruction is typically performed in infancy, untreated bladder exstrophy in adults is infrequent. An adult presenting with bladder exstrophy is quite uncommon. We present a 32-year-old man with a bladder mass that existed since birth. He complained of an unpleasant discharge from the mass upon presentation, and on examination, a mass was seen on the urinary bladder's exposed surface, coupled with penile epispadias, a deformed scrotum, and undersized bilateral testicles. Ultrasonography of the kidneys, ureters and urinary bladder (USG KUB), contrast-enhanced computed tomography (CECT) of the abdomen and pelvis, and mass biopsy were all used to investigate the patient. The patient was found to have signet ring adenocarcinoma of the urinary bladder. A radical cystectomy with an anterolateral thigh flap was performed. The clinical and radiological characteristics, treatments, and results of this uncommon presentation are discussed in this case report. Bladder exstrophy is uncommon, occurring 2.15 times per 100,000 live births , 4.We present the third case of signet ring cell adenocarcinoma in a 32-year-old man with untreated bladder exstrophy to highlight the extreme rarity, though a distinct possibility, and difficulties encountered in the surgical treatment of such patients.A 32-year-old unmarried man had had a bladder mass since birth. He had never consulted a doctor for the appearance of a lower abdominal mass and now presented with a foul-smelling discharge from the mass. On examination, there is an approximately 12 cm × 10 cm sized, an exophytic irregular mass that was non-tender and bled on touch and occupies the entire exposed urinary bladder surface , which was suggestive of signet ring cell adenocarcinoma under low Figure  and highBladder exstrophy is a rare disorder with an incidence of 2.15 per 100,000 live births [Only a very small portion of patients had bladder cancer that had not spread. A thorough literature search turned up fewer than 90 occurrences of primary bladder cancer in persons with untreated exstrophy. There are just two of these occurrences that are known to have their roots in India , 4.The possibility of developing cancer from untreated bladder exstrophy was originally identified in 1955 by McIntosh and Worley ,5. Two-tThe bladder mucosa in untreated exstrophy was comprehensively described by Smeulders et al. in 2001 ,6. ContrHistological subtypes of bladder adenocarcinomas might be mixed, enteric, mucinous, signet ring cell, or non-specific. In our situation, the histology proved that the cells were signet ring cells. Due to the rarity of this mutation, only one additional instance of signet ring cell cancer in an untreated exstrophy patient has been documented in the literature .According to McIntosh and Worley, the exstrophy bladder experienced glandular metaplasia as a result of environmental exposure and persistent infection, likely to provide protective mucus, where the malignant transformation took place . The intSuch situations are still typically treated with surgery. Adenocarcinoma and squamous cell carcinoma are examples of non-urothelial carcinomas for whom systemic treatment has not been effective ,9. The rLower abdominal defect correction after exstrophy bladder excision is challenging due to the typical large abdominal defect and widely separated rectus seen in these individuals due to pubic divergence. Therefore, the alternatives for repairing the defect are the rectus abdominal rotation flap, fasciocutaneous M-plasty, and Cardiff repair with onlay mesh repair -12.We preferred a pedicle of the anterolateral thigh flap based on perforators of descending branches of the lateral circumflex femoral artery.An adult seeking care for untreated exstrophy with primary bladder cancer is unusual but possible. Since the defect is so prominent at birth, nearly all patients undergo treatment soon after birth. Our patient never consulted a physician for the presence of a lower abdomen mass and presented to us with a foul-smelling discharge from the mass. Biochemical, radiological, and histopathological examinations helped in the proper evaluation of the mass.In our case, the histopathological examination revealed signet ring cell adenocarcinoma. We suggest radical cystectomy with an ileal conduit for these patients. With a pedicle or rotation flap, the substantial lower abdominal defect that was left open can be repaired. Therefore, a satisfactory functional and cosmetic outcome can be attained in bladder exstrophy patients who present late with good surgical care.


Inflammatory pseudotumor is a rare benign neoplasm that has been described in nearly the entire body and is often mistaken for malignancy. The exact etiology remains unknown. We present a case of small bowel obstruction secondary to an inflammatory pseudotumor. The patient’s symptoms and radiological findings were very concerning for underlying malignancy. En-block resection was recommended to prevent a recurrence. We aim to shed light on this rare cause of small bowel obstruction. Inflammatory pseudotumor is a benign neoplasm that clinically and radiologically mimics malignant tumors. The exact etiology remains unknown. It is presumed to occur secondary to infection, trauma, or surgery. The entire body can be affected, and it has a preference for children and young adults . The difA 53-year-old female presented to our emergency room with generalized periumbilical pain associated with nausea and vomiting. Her condition started two years prior with intermittent similar but less severe symptoms which were sometimes associated with diarrhea. Laboratory findings were within normal limits: sodium 136 mmol/L, potassium 3.9 mmol/L, creatinine 0.8 mg/dL, bilirubin 0.4 mg/dl, aspartate transaminase 74 U/L, alanine transaminase 16U/L, white blood cell count 9.3 K/µL, and hemoglobin 12.5 g/dL. Computerized tomography of the abdomen and pelvis revealed a high-grade small bowel obstruction in association with a heterogeneously enhancing lobular mass. The mass was partially encasing the small intestinal transition point and measured 42 × 65 mm, which was consistent with a neoplastic process, most likely small bowel lymphoma. A carcinoid tumor was also a possibility given her intermittent diarrhea symptoms  mimicking ovarian malignancy [In 1953, inflammatory pseudotumor was first described in a patient after right hepatic lobectomy . The etireaction . Inflamm culture . Mycobaclignancy . It alsoAlthough the lungs and orbit are most commonly involved, it has been reported in the liver, pancreas, mesentery, retroperitoneum, diaphragm, bladder, kidney, adrenal gland, esophagus, stomach, small intestine, colon, appendix, and Meckel’s diverticulum ,11.Morphologically, it is characterized by a mixture of myofibroblasts, fibroblasts, lymphocytes, and plasma cells in proportions varying from one microscopic field to another and from one tumor to another. Histologically, it contains cells associated with both acute and chronic inflammation, including lymphocytes and plasma cells, myofibroblastic spindle cells, and collagen .Radiological findings are variable on computerized tomography; the mass may be heterogeneous, homogeneous, and with no enhancement . The tumSpontaneous regression of a hepatic pseudotumor has been reported . High-doAdjuvant therapies consisting of cisplatin, doxorubicin, methotrexate, and radiation therapy have been used without great success . TreatmeInflammatory pseudotumor is a benign idiopathic inflammatory process with unknown etiology. It is misdiagnosed as an infectious or neoplastic process. It has been found and reported in almost every organ system. Clinical presentation is variable with nonspecific symptoms and symptoms related to the affected organ. Surgical en-block resection when feasible is recommended to obtain a histological diagnosis and minimize recurrence.


Malignant pancreatic tumors have early metastasis, aggressive behavior and poor prognosis. Surgeons often need to judge whether a patient needs prompt surgery when a pancreatic lesion is found. The accessory spleen is a congenital developmental malformation rather than a tumor and does not require surgical resection. Here, we report a 47-year-old man who underwent routine gastroscopic examination, and a submucosal eminence of the duodenal bulb was detected. The patient was asymptomatic and laboratory tests were unremarkable. Duodenal neuroendocrine neoplasm (G2) was considered following endoscopic submucosal dissection (ESD). Further examination showed a lesion in the tail of the pancreas and multiple accessory spleens. The lesion in the tail of the pancreas was Ga-68 positive and was highly considered a pancreatic neuroendocrine tumor (pNET). Based on this clinical evidence, laparoscopic spleen-preserving distal pancreatectomy (Kimura) was performed. However, the results of the postoperative pathological diagnosis indicated an intrapancreatic accessory spleen (IPAS). Given the findings of this case, we should explore more accurate diagnostic methods for IPAS to avoid unnecessary surgery. Accessory spleen (AS) is a congenital developmental malformation that mainly occurs in the splenic hilum 62.1%) and tail of the pancreas 5.5%) % and tai. Researc.5% % andAdvances in imaging diagnosis technology have helped identify an increasing number of people with pancreatic masses and, simultaneously, AS. ASs located in the pancreas are defined as intrapancreatic accessory spleens (IPASs). IPASs usually show no obvious symptoms and are often discovered in routine radiology examinations. There are no significant radiological differences between IPASs and pancreatic neuroendocrine tumors (pNETs), which may lead to a misdiagnosis. Here, we discuss methods for accurately diagnosing IPAS through a case encountered in our center.A submucosal eminence of the duodenal bulb was discovered in a 47-year-old male patient after agreeing to undergo routine electronic gastroscopy in a local hospital. The eminence measured approximately 3*3 cm and presented with hyperemia on its surface. The patient had no chest tightness, asthma, bloating, abdominal pain or other symptoms. Ten days later, he was transferred to another hospital and underwent an abdominal enhanced CT scan as well as endoscopic submucosal dissection (ESD). The pathology results after ESD indicated that the mass was a neuroendocrine tumor (NET), the immunohistochemical report showed CgA (+), Syn (+), CD56 (+/-), SSTR2 (+), and Ki67 (4%+), and the mitotic count was 0-1/10 high-power fields (HPF). The mass was diagnosed as a NET-G2.Given the malignant and metastatic potential of NETs and that the pathological grade of metastatic lesions may be higher than that of primary lesions, the patient underwent Ga-68 examination at a third hospital to evaluation whether other NETs were present in the abdomen and the rest of the body. Imaging revealed a high-uptake nodule in the tail of the pancreas and three accessory spleens with low uptake around the pancreas 2For further treatment of the pancreatic tail lesion, the patient was transferred to our hospital. Neither his physical nor experimental examinations showed remarkable disorders. The patient’s serum tumor markers  and carcinoembryonic antigen (CEA)) were normal. After admission, endoscopic ultrasonography (EUS) was performed. We found a quasicircular, slightly hypoechoic lesion measuring approximately 10*8 mm. The lesion was located in the tail of the pancreas, close to the splenic hilus. To further clarify the nature of the lesion and uncover other lesions, we performed dual-energy CT (DECT) of the pancreas before continuing with the next treatment steps. From the CT scan, we observed a nodular soft tissue density shadow in the tail of the pancreas measuring approximately 10*11 mm. It was significantly enhanced in the arterial phase, weakly enhanced in the venous phase and had a slightly low density in the delayed phase. Additionally, there were multiple soft tissue density shadows around the spleen, which were considered small ASs Figure 3The diagnosis of pNET by Ga-68 PET/CT mainly depends on the high expression of SSTR (mainly somatostatin receptor 2 (SSTR2) and 5 (SSTR5)). According to the earlier PET/CT results of this case, the lesion of the pancreatic tail was Ga-68 positive, indicating a highly likelihood of a misdiagnosis of NET. Therefore, we further performed additional immunohistochemical experiments. The SSTR results were SSTR2(+) and SSTR5(-) Figure 4In this case, an IPAS was misdiagnosed as pNET, resulting in unnecessary surgery. However, the patient underwent relatively sufficient examinations before the operation including radiography, endoscopy and PET/CT imaging. The preoperative examination results did not suggest the possibility of a diagnosis of IPAS, which led to the implementation of unnecessary surgery. Given the results of this case, we attempted to determine the causes of the misdiagnosis and identify better differential diagnosis methods.via endoscopic ultrasound-fine needle aspiration (EUS-FNA) biopsy  biopsy . IPAS cavia EUS or ESD is often needed. At present, a variety of markers are used in the pathological differential diagnosis of IPAS and pNET, such as CK-pan, Vim , CgA, Syn, CD56 (NET diagnostic markers) and Ki-67 (grading markers). It is worth noting that CD8 has been reported to be more conducive in improving the accuracy of the differential diagnosis between IPAS and pNET  for the publication of any potentially identifiable images or data included in this article.ZYH, SGD and MLD conceived the case and wrote the manuscript. WJ reviewed pathological results. HQQ interpreted the PET/CT results. ZK, DX and LZP interpreted CT results. WJL and JKR helped revise and polish the manuscript. All the authors contributed to the article and approved the submitted version.Our work was supported by the National Science Foundation for Young Scientists of China (No. 81902455); the National Natural Science Foundation of China ; Jiangsu key Medical Discipline .All the authors thank the members of their research group for useful discussions.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.


A pleomorphic adenoma or mixed tumor is a heterogeneous benign tumor of the salivary glands. The most frequent site is the parotid gland. It is rare in the accessory salivary glands, preferably located in the oral mucosa . The diagnosis of pleomorphic adenoma of the palate can only be made on a biopsy while remaining vigilant about the possible existence of other neoplastic foci within it. Definitive anatomopathology after excision is mandatory. Here, we present a clinical case of pleomorphic adenoma of the palate. We propose to specify the epidemiological, diagnostic, and therapeutic particularities of these tumors as well as their evolutionary characteristics. The palatal vault is the site of a wide variety of tumors. Most are benign, some are true cancers, and others are difficult to classify and evaluate. The accessory salivary glands, spread throughout the upper aerodigestive tract, can be the site of tumors, mainly in the mucosa of the mouth , more rarely in the larynx or nasal cavity. The tumor varieties are numerous; the most common tumors that originate from the minor salivary gland are malignant tumors and benign tumors that recur in rare cases and can transform into carcinoma after a long evolution .A 26-year-old female patient, with no specific pathological history, consulted in September 2021 for a swelling of the left soft palate. The swelling started three years earlier with the appearance of a small nodule that progressively increased in volume to fill the entire left hemipalate. Clinical examination revealed a swelling of the left hemipalate, 31 × 32 mm in diameter, firm, painless, not bleeding on contact, and lined by normal mucosa. There was no cervical adenopathy or other palpable cervicofacial mass. Computed tomography (CT) scan performed before and after injection of contrast medium showed a 32-mm mass of well-limited tissue appearance of the soft palate lateralized to the left but discreetly protruding from the median line of regular contours of non-univocal nature  showed a voluminous lesion appearing at the junction between the left soft palate and the palatine tonsil measuring 28 × 26 × 27 mm evoking a pleomorphic adenoma Figures . A biopsThe resection was considered total intraoperatively with a tight closure of the mucosa Figure  after plThe histological examination of the surgical specimen showed an encapsulated tumor proliferation with a double contingent: a connective contingent made of spindle cells without cytonuclear atypia and an epithelial contingent made of tubes and trabeculae of cubic cells without cytonuclear atypia Figure .We concluded that it was a pleomorphic adenoma. Feeding was provided for three days by a nasogastric tube. Postoperative care consisted of antibiotic therapy and local betadine care. Postoperative follow-up was simple. Control at 10 months of evolution did not reveal any local recurrence.Tumors of the accessory salivary glands are very rare. The palate, with more than 50% of the accessory glands equally divided between hard and soft palate, represents the preferential site of accessory salivary tumors (two-thirds of cases). Pleomorphic adenoma of the roof of the mouth or soft palate occurs on average between the third and fifth decade of life and affects slightly more women than men ,2.Salivary tumors of the palate are characterized by discreet functional signs: slight discomfort when swallowing, and sensation of a foreign body. The palatal mucosa is respected for a long period only appearing blown by the tumor formation. They are generally firm and hard to palpation. At the exulceration stage, the tumor often appears grayish-white with necrotic areas . SeveralThe definitive diagnosis is essentially based on the anatomical-pathological examination of the surgical specimen. Histologically, the pleomorphic adenoma presents as islands of spindle cells on a myxoid background, an internal layer of epithelial cells, and an external layer of myoepithelial cells. Three main subtypes have been identified, namely, myxoid (stroma accounts for 80%), cellular , and mixed  . The latThe treatment of pleomorphic adenoma of the palate is based on wide surgical excision to avoid the risk of recurrence ,6.Tumors of the palate are rare and of various histological types. The possibility of a pleomorphic adenoma must be evoked in case of palate swelling. The literature confirms that this tumor is progressive and invasive in case of surgical abstention and it tends to recur locally and regionally. Complete removal is imperative. Regular locoregional surveillance after excision must be systematic for several years.


Testicular torsion is a relatively common urological emergency, which involves the twisting of the spermatic cord and its contents leading to ischemia to the testes, which usually presents as sudden, severe scrotal pain. In comparison, testicular neoplasms are far less commonly encountered in the emergency department as they often present as painless hard masses that grow slowly over longer periods of time. Extremely rare cases of testicular neoplasms present as sudden scrotal pain that causes a challenging task in the emergency department as physical examinations and ultrasound findings could vary and not be specific enough in confirming the diagnosis.In this case, we report a 22-year-old male who was referred from the emergency department (ED) as a case of testicular torsion from the presenting history; however, his physical examination and Doppler ultrasound findings were suspicious of testicular malignancy. The patient presented with a history of right scrotal pain for a few hours with no predisposing factors; however, examination and imaging were highly suspicious of an underlying neoplasm. The patient underwent an inguinal orchidectomy, and histology confirmed the presence of a germ cell tumor of varying components.In conclusion, a high index of suspicion for testicular torsion should always be present when a patient presents with sudden onset testicular pain; however, the differential diagnosis including testicular neoplasms should not be overlooked as it can change the management and outcome. The annual incidence of testicular neoplasms is around two to three cases per 100,000 male population in the United States . HoweverIn contrast to testicular cancer, testicular torsion is a relatively common urological emergency with the incidence reaching one in every 4000 males younger than 25 years in the United States per annum . It invoIn this case, we report a 22-year-old male who was referred from the emergency department (ED) as a case of testicular torsion from the presenting history; however, his physical examination and Doppler ultrasound findings were suspicious of testicular malignancy.A medically fit 22-year-old male presented to ED with a history of right testicular pain for a duration of four hours. The patient reported that the pain had started acutely with no predisposing factors or trauma. Upon scrotal examination, the right hemiscrotum appeared to be swollen and erythematous with the right testis being extremely tender to touch. In addition to that, a hard mass was also palpable, and this is what initially raised the possibility of an underlying malignancy versus the initial impression of testicular torsion. Furthermore, his initial investigative workup including a full blood count and urine analysis was unremarkable.A color Doppler ultrasonography (CDUS) was arranged urgently and revealed a heterogenous appearance of the right testis with no evidence of normal testicular tissues as well as multiple macrocalcifications and cystic changes; however, no evidence of torsion of the right spermatic cord was noted Figure .Once these findings were observed, the diagnosis of a testicular neoplasm was highly suspected. Consequently, the condition was discussed in detail with the patient and his family, and they were informed of the possibility of an underlying testicular neoplasm. The patient was informed that he will require surgery for testicular exploration with a high possibility of right orchidectomy through an inguinal approach.Moreover, prior to surgery, blood tumor markers were sent and showed an elevated alpha-fetoprotein (AFP) of 136.9 µg/L, beta-human chorionic gonadotropin (beta-HCG) of 942.1 mIU/mL, and lactate dehydrogenase (LDH) of 555 U/L adding to the diagnosis of an underlying testicular tumor. The patient was taken for right inguinal orchidectomy, and intraoperatively, the right testis appeared as a hard mass without twisting of the spermatic cord, hence confirming the absence of testicular torsion. The spermatic cord was ligated near the internal inguinal ring, and an inguinal orchidectomy was performed with the right testis sent for histopathology review.The patient was discharged on postoperative day 2 in a stable condition. The right testicle histology report exhibited a mixed germ cell tumor with components described as 50% embryonal carcinoma, 35% teratoma, and 15% yolk sac tumor with lymphovascular invasion. Following histological confirmation of the diagnosis, a staging computerized tomography (CT) scan of the chest, abdomen, and pelvis was done and denoted pulmonary metastasis with extensive involvement of the retroperitoneal and common iliac lymph nodes (Figure Moreover, there was no evidence of bone or liver metastasis. Given this information, the staging of this patient’s neoplasm was regarded as T2N2M1. Finally, the case was presented in the National Tumor Board multidisciplinary team meeting and referred to the medical oncology team for further management.Acute testicular pain in all prepubertal and young adult males should be considered as testicular torsion until proven otherwise. This makes prompt diagnosis and management to be of paramount importance since earlier time to surgical intervention is directly proportional to future testicular viability . In compThe recognized presentation of testicular neoplasms is usually contradictory to that of testicular torsion, in which the earlier presents as a slow-growing painless mass over months and the latter as a sudden severe scrotal pain over hours  adding tIn addition to that, it has been reported that color Doppler ultrasound imaging has a high sensitivity 100%  and specIn conclusion, as illustrated in this case, proper physical examination and CDUS aided in early recognition of the underlying testicular neoplasm rather than testicular torsion, and this prevented immediate scrotal exploration through a scrotal approach; however, as seen across the sparse literature regarding similar cases, the diagnosis can be easily missed.


Salivary gland tumors can also occur rarely in the retromolar area, though common near the junction of hard and soft palate, labial mucosa, and buccal mucosa. Most salivary gland tumors in the retromolar pad area are malignant and should be excised. The cystadenoma is a rare, benign, salivary gland tumor. Importantly, incomplete resection of this tumor can lead to recurrence or cervical lymph node metastasis. We reported herein a case of cystadenoma arising in the right retromolar pad area in a 63-year-old male patient who underwent reconstruction using a buccal fat pad flap (BFPF) after the surgical removal of the tumor with a 10-mm margin left a defect with bone exposure. No evidence of recurrence or complication was found at the postoperative, three-year follow-up. Salivary gland tumors are relatively rare in the oral cavity but can potentially affect the minor salivary glands significantly. Minor salivary gland tumors comprise fewer than 25% of salivary gland tumors . The cysThe buccal fat pad flap (BFPF) is a reliable and straightforward method of treating intraoral soft tissue defects. It has been used to reconstruct maxillary defects induced by tumors since it was first reported in 1977 [A 63-year-old male patient was referred by his dentist to our hospital to treat an asymptomatic mass in the right retromolar pad area, which was discovered incidentally during dental treatment. There was no history of oral surgery and oral trauma. The patient had no history of smoking or alcohol consumption and no exposure to environmental carcinogens. Clinical examination revealed a mass of 17 mm x 10 mm without tenderness and a reddish area with discrete papules bordered by a peripheral whitish area on the lingual aspect in the right retromolar pad area , suggestive of secondary caries with a chronic periapical abscess in the mandible Figure .Contrast-enhanced magnetic resonance imaging (MRI) with short tau (TI) inversion recovery demonstrated a hypointense lesion with a sharp margin, round shape, and uniform density distribution lesion in the right retromolar area, which was 13 mm x 10 mm in size and had not invaded the surrounding tissue  was administered until the day after surgery. The patient began a dysphagia soft diet the day after surgery. Mouth opening exercises were started in the early postoperative period. The postoperative course was uneventful, and the patient’s mouth opening was unconstrained. No recurrence was found at the three-year follow-up Figure .We reported a rare case of cystadenoma in the retromolar pad area. It should be borne in mind that a neoplastic lesion developing in the retromolar pad area may be a malignant salivary gland tumor. Therefore, in the present case, we performed a wide resection and reconstructed the surgical defect using a BFPF with good outcomes.Cystadenomas, which arise in the pancreas, ovary, prostate, or kidney -11, rareIn general, it was predominantly found in females . Table 1The wide excision in our patient left a defect with mandibular bone exposure in the retromolar pad area, which was corrected using a BFPF. The BFPF has the advantage of being simple and easy to perform. The BFP consists of the main body with four extensions: the temporal, buccal, pterygoid, and pterygopalatine processes . GeneralAccording to a review of 12 studies, 89.1% of BFPF reconstructions had no associated adverse events, including infection, loss of graft, bleeding, fistula, dehiscence, and mouth opening limitation . HoweverIn this case, the biopsy result was sialadenoma papilliferum. However, the resected specimen was finally diagnosed as a cystadenoma. Sialadenoma papilliferum usually occurred at the junction of the hard and soft palates and the floor of the mouth and histopathologically did not show multiple cystic cavities . The hisMost salivary gland tumors in the retromolar pad area are malignant . AlthougWe experienced a case of cystadenoma occurring in the retromolar pad area, which was widely resected. The surgical defect was reconstructed using a BFPF. Most salivary gland tumors in the retromolar pad area are malignant, but a benign tumor may occasionally occur, as in the present case. Nonetheless, even a benign salivary gland tumor, especially a cystadenoma, should be excised completely to prevent a recurrence.


We present a case of stage IV uterine leiomyosarcoma that was treated with total hysterectomy, bilateral salpingo-oophorectomy, and extensive resection of a 14 kg abdominal mass, as well as complete eradication of accompanying symptoms. This case may prompt researchers to look for other surgical solutions to similar issues. •Stage IV Uterine Leiomyosarcoma can be surgically curable.•Total Hysterectomy and Bilateral Salpingo-Oophorectomy is the surgical choice in massive Uterine Leiomyosarcoma.•Post-operation Follow-ups with Ultrasonography at a 3-months intervals are crucial to detect early relapse. LMS accounts for about a quarter of all soft tissue sarcomas, and the incidence depends on gender, subtype, and location . TheoretLMS staging and grading systems are commonly used to create risk profiles. However, ULMS itself contributes to a poor prognosis, even if it is confined to the uterus. Currently, the revised criteria of the International Association of Obstetrics and Gynecology (FIGO) is superior to other classification systems in terms of progression and overall survival but is not ideal ,6. ULMS Surgery is the main treatment option. In general, aggressive surgical cytoreduction at the time of initial diagnosis offers the possibility of prolonging survival or healing . InitialWe present a case of high-grade stage IV ULMS. Hysterectomy and bilateral salpingo-oophorectomy were performed and 14 kg of abdominal masses were resected. It was then followed by subtotal resolution of the tumor's symptoms The manuscript has been reported in line with the SCARE 2020 criteria .2We present a 53-years-old multiparous nonsmoker female.. Her pharmaceutical, familial, and medical histories were all uncomplicated. Her surgical history included only two cesarean sections.She presented to her local clinic two years ago with vague abdominal and lumbar pain. Her pain was unrelated to posture or dietary habits. No aggravating or alleviating factors were reported.During clinical examination, mild discomfort was noted in the left upper quadrant during palpation. Routine blood work and U&E were within normal limits. An abdominal ultrasound revealed a mass in the left upper quadrant and some irregularities in the abdomen, but without proper details. CT imaging has been ordered and the results were:2.1Observation of (7*10) cm mass in the medial basal left chest. Observation of small multiple bilateral masses in the lung parenchyma. Observation of minimal bilateral pleural effusion.2.2Observation of wide-spread multiple necrotic lesions; the biggest of those measures (25*30) cm.Later, a needle biopsy of the chest mass has been ordered. Microscopic pathology revealed the presence of a few macrophages with focal small alveoli, fibrotic walls, chronic inflammatory infiltrate, and anthracosis. This pathology was consistent with hepatic cell carcinoma, but immunostaining showed Chromogranin positivity and on this basis, a diagnosis of the moderately differentiated neuroendocrine tumor was made. As a result, it was initially placed on Octreotide.After 4 months, MSCT was ordered for follow-up. It results in slight development of the tumor as follows:2.3The (7*10) cm mass in the medial basal left chest has started stretching to the posterior mediastinum. The multiple bilateral masses showed no signs of retraction. Lymphadenopathy in the mediastinum and the axilla see .Fig. 1Ut2.4A huge, space-occupying mass begins to march up from the pelvis until it reaches the diaphragm. It's pushing relevant relationships through its course. It includes macrocalcifications and multinecrotic liquefied hypoechoic areas shown in . LymphadAt this point, the patient has unintentionally lost more than 15% of their body weight since the first presentation. She reported that her appetite was significantly reduced due to vague general abdominal pain and persistent vomiting. She also suffered from constipation and painful bowel movements. After several discussions with general surgery, she preferred to forgo the palliative solution and to perform an excisional operation. Based on this, elective laparotomy was planned.After proper examination of the abdomen and pelvis, total hysterectomy and bilateral salpingo-oophorectomy were decided. In addition, a total of 14 kg of mass was removed from the abdomen, allowing decompression of the small intestine, large intestine, stomach, and kidneys. Lung lesions were left without intervention Shown in . PatholoAfter more than a year of surgical resection, she reports happiness with full resolution of abdominal symptoms. She has regained her weight. She is on multiple courses of Gemcitabine and Vinorelbine. Her follow-up b laboratory tests show no abnormal findings other than occasional leukocytopenia that can be explained by chemotherapy. According to follow-up CT, lung lesions are showing small or unnoticeable retraction. No respiratory symptoms are reported at the moment.3This ULMS is a stage IVB according to FIGO. Some conservative surgical treatment is offered, yet it's not traditionally treated with wide excision surgery. However, nothing in the literature -to the extent of our knowledge-goes completely against it.The main purpose of excisional surgery was to decompress the abdominal structures. The purpose was achieved as she regained her weight with normal eating and bowel movements. The uterus played an obscure role in the lesion prior to surgery.MRI was not possible at the time due to the lack of hospital equipment. Total hysterectomy and bilateral salpingo-oophorectomy were decided after intraoperative inspection of the uterus and ovaries. We concluded that the primary tumor was ULMS. The metastases and the deposits in the abdomen were likely the cause of the first complaints of abdominal pain. A later resection operation for lung metastases should be planned.It is known that uterine sarcomas spread via both lymphatic and hematogenous dissemination. Similar to our case, distant metastasis to the lung can frequently happen. Ovarian metastasis is considered rare . In Fig,Ultrasonography is being repeated at a 3-months interval as it’s more reliable for postoperative follow-up than preoperative diagnosis . No recuWe do not know whether the premenopausal status causes a good prognosis because the postmenopausal status is associated with histological types of poor prognosis . No abnoThis case shows adequate surgical resolution of the Stage IV ULMS primary tumor. Although it is common practice for Stage IV cancer not to be treated surgically, we recommend further studies along with this case to discuss and revise current guidelines.The case report conducted ethically in accordance with the World Medical Association Declaration of Helsinki. The study was approved by Ibn Al Nafees Hospital Committee, decision number N1178 dated 04/05/2021. The patient has given their written informed consent to publish their case (including publication of images).This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.All authors have participated in writing and reviewing the manuscript.Not applicable.Mohammad Badr Almoshantaf.The data that support this case report is not publicly available due to privacy purposes but are available from the corresponding author upon reasonable request.Not commissioned, externally peer-reviewed.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.The authors have no conflicts of interest to declare.


Von Recklinghausen’s disease is characterized by the presence of cafe-au-lait spots, multiple neurofibromas and hamartomas. It is a dominant autosomal disease with high penetrance and variable expression, involving the head and neck in 22 to 47% of the patients. It is known as type I neurofibromatosis, in order to differentiate it from central involvement, especially that of the eight cranial nerve (acoustic neuroma - type 2 neurofibromatosis)Malignant transformation varies between 2% and 40%, and these patients are more prone to having malignant tumors of the nervous tissues and other secondary neoplasiasMalignancy must be suspected when there is a fast growing and painful mass, investigated by image exams  for staging, resection evaluation and lesion biopsy purposesClinical development is characterized by local recurrences, and has poor prognosis in patients with multiple neurofibromatosis, and the lungs are frequently the seat of distant metastasisTreatment is based on radiotherapy, chemotherapy and surgery; it bears a low 3-year survival rateC.E.R., 37 years, single, Caucasian, with previous diagnosis of von Recklinghausen’s disease and a history of a fast-growing mass in the right supraclavicular fossa for one year . PhysicaMicroscopy: malignant mesenchymal tumor made up of ovoid and spindle-like core cells, coated by an elongated cytoplasm, creating multidirectional bundles, with a moderate mitotic index, with hypercellularity and necrotic areas.Diagnostic assumption: high grade spindle-cell sarcoma. Submitted to radiotherapy. The patient developed pain and a nodule in the right neck region. A new CT scan indicated lesion without vascular invasion and a nodule near the lung dome (probable recurrence). Submitted to palliative chemotherapy.U.G.P., 39 years old, Caucasian, neck tumor for 6 months; we noticed a neck enlargement with bilateral masses that pushed the larynx to the right, almost extreme lateral. A patient with von Recklinghausen’s disease already submitted to resection of nodules in the chest and left shoulder. MRI showed a large solid neck tumor on the left side - compressing vessels, the larynx and trachea to the right side, all the way to the upper and anterior mediastinum, and large neck neuromas, larger on the left side. Nasal fibroscopy showed left vocal fold paralysis. The patient was taken to palliative surgery, in an attempt to decompress the trachea and avoid invasion of the large vessels. Macroscopically the lesion had 15.5 cm and weighed 350g. Histopathology: high grade spindle-cell sarcoma . Immunohistochemistry test showed sarcoma.Pain and fast growth associated with neurofibromas leads us to suspect of sarcomatous transformation, although it can be a benign lesionThe cases reported are of patients with von Recklinghausen’s disease, aged between 37 and 40 years, males, non-smokers, with a fast growth lesion in the neck and supraclavicular regions.Diagnostic investigation was carried out by image exams  and histopathology.Surgery was carried out, resecting the lesions and involved adjacent structures, added to adjuvant radio and/or chemotherapy.Both cases were diagnosed and treated in a late stage, thus, unfortunately being of palliative nature only.These patients must be continuously followed up because of the possibility of the patient developing metastatic disease, especially in the lung, and have local recurrence.


The falcine sinus is a normal embryonic structure that is situated between the 2 layers of the falx cerebri and drains the deep cerebral venous system into the superior sagittal sinus. It normally involutes after birth and is uncommon in adults. Although it is often an isolated and incidental finding, it can also be associated with a number of other conditions including but not limited to vein of Galen arterial malformations (VGAM), atretic parietal cephaloceles, acrocephalosyndactyly (Apert syndrome), absence of the corpus callosum, absence of the tentorium, osteogenesis imperfecta, or Chiari II malformations. We present a case series of 3 pediatric patients born with a persistent falcine sinus and an associated condition, including a VGAM, an APC, and a sinus thrombosis. The purpose of this article is to highlight the importance of understanding anatomic variations in the cerebral venous system to help aid in the proper diagnosis and treatment of associated pathologies. The falcine sinus is located in the falx cerebri and drains the posterior vein of Galen of the deep cerebral venous system to the superior sagittal sinus in the fetus . It normDuring embryonic development, the primitive falx cerebri contains a mesh of anastomotic venous loops known as the sagittal plexus. One of the caudal anastomotic loops of the sagittal plexus gives rise to the falcine sinus, which connects the posterior vein of Galen with the superior sagittal sinus in the developing fetus. The falcine sinus normally disappears after development of the superior sagittal sinus and straight sinus are complete Persistent falcine sinus is not an uncommon incidental finding in pediatric patients. In a retrospective review of 3135 pediatric brain MRIs completed at Penn State Hershey Medical Center between January 2008 and December 2009, 1% of patients were found to have a persistent falcine sinus In her first week of life, a baby girl born with a history of VGAM discovered in utero presented for a follow up neonatal head ultrasound. Ultrasound redemonstrated the VGAM, as well as a persistent falcine sinus . Same daInterventional embolizations were performed in stages to reduce the risk of thrombosis or hemorrhage. By 21 months old, the patient underwent 6 arterial embolizations, which appeared to be curative. As of age 5, the patient appears to be doing very well developmentally and intellectually. She has no cognitive or neurological problems and is meeting all developmental milestones.A baby boy was born at term by normal vaginal delivery with neonatal jaundice and a 2 cm midline, parietal, skin-covered, violaceus, non-pulsatile mass . Head USAt 3 months old, the mother reported a possible “leak,” so the APC was excised earlier than initially planned. The APC was opened following resection and was found to be free of any structures. Its stalk was about 5 mm in external diameter and its lumen was very tiny. The patient tolerated the procedure well. At 3 years old, the patient underwent a cranioplasty to repair his skull defect. The patient also has a history of aortic root dilation, autism, and strabismus associated with chromosome 7q11.23 duplication syndrome.A baby boy was born at term via C-section for breech presentation. He was tachypneic with a “high pitched cry” shortly after birth. Out of concern for sepsis, a diagnostic lumbar puncture was performed demonstrating gross blood. Neonatal head US evaluation revealed a large falcine sinus. At approximately 36 hours of life, a follow up head CT  and braiThe posterior branches of the pericallosal arteries normally anastomose with the distal branches of the posterior cerebral arteries before birth. In VGAMs, these anastomoses degenerate, leading to an enlarged and malformed MPV . The decCephaloceles are rare herniations of intracranial structures through a congenital bone defect. They have an incidence of between 1 in 3500 and 1 in 5000 live births. Of these, 37.5% are atretic cephaloceles which are cephaloceles comprised of dura, fibrous tissue, and dysplastic brain tissue Neonatal cerebral venous sinus thrombosis is rare, and associated with comorbid conditions, 62% of the time, including dehydration (26%), sepsis (7%), cardiac defects (26%), and meningitis (10%) The persistent falcine sinus may be associated with a diverse array of anomalies and is not an uncommon incidental finding. Although commonly seen with VGAM, arteriovenous malformations, absent corpus callosum, osteogenesis imperfecta, or Chiari II malformations, in this study, it was seen with VGAM, atretic parietal cephalocele, malformations of cortical development, and sinus thrombosis. We highlight the need to understand anatomic variations and anomalies in the cerebral venous system to ensure the proper diagnosis and treatment of the associated pathology.Appropriate informed written consent has been obtained for this publication.


Neurofibromatosis type 1 is characterized by multiple cutaneous neurofibromas of varying sizes along with skeletal, neurologic, and ophthalmic features. Solitary swellings in neurofibromatosis type 1 are not commonly encountered except in the form of plexiform neurofibromas. We report two cases with neurofibromatosis type 1 presenting with solitary swelling in the ankles which were proven to be the diffuse type of neurofibroma, radiologically and histopathologically. Diffuse type neurofibroma presenting as ankle swelling in type 1 neurofibromatosis has not been reported before. Neurofibromatosis type 1 or von Recklinghausen disease is an autosomal dominant genodermatosis presenting with multiple cafe-au-lait macules, single or multiple neurofibromas, axillary and inguinal freckling, Lisch nodules, and skeletal and central nervous system abnormalities . SolitarCase 1An 18-year-old female presented in the department of dermatology in a teaching hospital in Chennai with the chief complaint of diffuse swelling of the right ankle for the past eight years. The swelling was insidious in onset, gradually increased in size, and was not associated with pain. There was a history of multiple pigmented lesions over the trunk since birth. There was no history of similar swellings elsewhere in the body and no history of central nervous system involvement such as seizures, visual disturbances, or hearing impairment. There was no positive family history, she was born of non-consanguineous parents and her developmental milestones were normal.On examination, a single irregular swelling of size 15 cm × 11 cm was present encircling the right ankle. The swelling was soft in consistency and not tender on palpation Figures , 1B. It Numerous pigmented macules were present bilaterally over the cheeks and lips Figure  along wiMultiple hyperpigmented macules noted to be as freckles were seen in both palms Figure . The patOn investigations laboratory parameters were normal. A biopsy was taken from the swelling and histopathology revealed a normal epidermis, with the dermis showing an unencapsulated tumor composed of spindle cells with elongated, wavy nuclei regularly spaced among thin wavy collagenous strands merging into the adjacent fascia Figures , 5B.Skiagram of the affected limb showed soft tissue shadows around the ankle joint with slight bowing of the tibia Figure . MRI of Case 2A 24-year-old male presented in the dermatology department in a teaching hospital in Chennai with a swelling of the left ankle for five years which was insidious in onset and was gradually increasing in size Figure . There wThe general examination was normal and vital signs were stable. There was no neurological deficit. The patient had multiple café-au-lait macules of varying sizes ranging from 2 cm to 5 cm over the back and inguinal region Figure . He alsoTwo Lisch nodules found in the right eye were confirmed by the ophthalmologist Figure . This paIn this patient, an 11 cm × 7 cm subcutaneous swelling was also present over the medial aspect of the left ankle obscuring the medial malleolus. The swelling was uniformly soft without the irregular bag of worms consistency and was not tender. The peripheral nerves were not thickened or palpable.A biopsy was done from the swelling that exhibited features of neurofibroma with the dermis showing an unencapsulated tumor of cells with an oval or spindle-shaped nucleus and scant, indefinite cytoplasm, merging imperceptibly to the surrounding tissue Figure . A cliniThe diagnostic criteria for NF1 are met in an individual who does not have a parent diagnosed with NF 1 if two or more of the following are present: though both patients did not have a parental history of neurofibroma, they fulfilled three of the diagnostic criteria of the revised diagnostic criteria for NF 1 as seen in Table Neurofibromatosis type 1, according to Riccardi's classification, is an autosomal dominant inherited syndrome manifested by developmental changes in the nervous system, bones, and skin. In type 1 neurofibromatosis, also called Von Recklinghausen’s disease, patients have multiple neurofibromas, cafe-au-lait spots, axillary freckling, and other skeletal and ophthalmic manifestations. Diagnostic criteria for NF1 have been defined taking into account these features . Most ofClassical neurofibromas are benign tumors that present as cutaneous tumors of varying sizes, numbers, and morphology. They rarely occur before late childhood, but continue to develop throughout life. These tumors usually arise from the nerve sheaths of the peripheral nerves or cranial nerves varying in number from a few to hundreds. They can be invaginated, as if through a ring in the skin, by pressure with the finger, called the buttonhole sign. Plexiform neurofibromas present as large, irregular bag-like swellings with the typical bag of worms appearance and occur along the course of a nerve .When neurofibromas present as solitary tumors they are usually seen as localized plexiform neurofibromas as stated in Table Diffuse neurofibromas usually manifest as diffuse dermal plaque-like swellings in the head and neck regions resulting in enlargement of the affected tissue . These aOn investigations, skeletal x-ray of the affected limb in both patients showed soft tissue shadows around the ankle joint with a slight curvature of the tibia in one of the patients. MRI of the ankle swelling revealed a T1++ hypointense to T2 hyperintense lesion in the posterolateral aspect of the ankle. These findings were similar to the MRI findings of diffuse neurofibromas reported in the literature . In addiHistologically, diffuse neurofibromas are described to be unencapsulated spindle cell tumors with bland cytologic appearance, serpiginous nucleo-cytoplasmic contours, and poorly delimited boundaries with the surrounding dermis . This waNerve sheath tumors such as neurofibromas and schwannomas are quite rare around the foot and ankle accounting for 5.4% and 3.9%, respectively, of all benign soft tissue tumors in this region . UsuallyDiffuse solitary neurofibromas can lead to complications such as limb hypertrophy and dysfunction. Unilateral hypertrophy in an extremity not only involves soft tissue hypertrophy but can also infiltrate the long bone cortex with narrowing of the medullary canal with cortical thickening . In addiSolitary neurofibromas not associated with NF 1 rarely become malignant, but when associated with the NF1 syndrome, they has the potential for malignant transformation. It has been reported that 12% of patients with neurofibromatosis type 1 will eventually develop malignant nerve sheath tumors from the neurofibromas of the oral cavity . SolitarUnilateral enlargement of the ankle swelling causing cosmetic and functional deformity and the risk of potential malignant transformation led us to refer our patients to plastic surgery where the swellings were excised successfully with a good cosmetic and functional outcome.This report of two cases with the rare presentation of solitary diffuse neurofibromas around the ankle joint in NF1 emphasizes the awareness that clinicians should have in encountering unusual manifestations of common genodermatoses.Arriving at the right but rare diagnosis among the spectrum of differentials in unilateral ankle swelling involves good history taking and an astute clinical examination. In our patients, pigmentary lesions such as café-au-lait macules, axillary and palmar freckling, and Lisch nodules helped us to arrive at the diagnosis of NF1 without the typical multiple cutaneous neurofibromas. Biopsy of the solitary ankle swelling established the diagnosis of diffuse neurofibroma in addition to the radiological findings. We recommend that this unusual variant of NF1 presenting as solitary diffuse neurofibroma around the ankle joint be included among the various clinical manifestations of neurofibromatosis type 1.


Duodenocolic fistula is a rare upper gastrointestinal fistula that can be benign or malignant. However, benign duodenocolic fistulas are particularly rare. Duodenocolic fistulas are often a complication of advanced colon cancer. The most common cause of benign fistulas is perforation of the duodenal ulcer. We report a case of a benign duodenocolic fistula in a patient who presented with abdominal pain, diarrhea, and weight loss. Gastroscopy and an upper gastrointestinal study confirmed the presence of the fistula. Surgery was performed, and the pathological examination demonstrated that the fistula originated from a duodenal ulcer. Duodenocolic fistula is a rare type of upper gastrointestinal fistula. Perforated duodenal ulcers are usually the most common cause of benign fistulas . MalignaA 53-year-old man presented with a 20-year history of abdominal pain, nausea, vomiting, and one episode of hematemesis. In April 2012, the abdominal pain was aggravated and was accompanied by edema of the lower extremities and abdominal bloating. Subsequent gastroduodenoscopy confirmed a duodenal ulcer. He was infected with Helicobacter pylori. The patient was treated with amoxicillin, clarithromycin, and protection of the gastric mucosa. Albumin infusion and diuretics were administered for hypoproteinemia and ascites. The patient's clinical condition had greatly improved and he was asymptomatic for 9 years. In November 2021, he developed abdominal pain and diarrhea and experienced the occasional passage of undigested food, general malaise, and slight edema of the lower limbs. He also experienced weight loss of 10 kg over the course of 6 months.Examination revealed normal vital signs; however, the patient was thin and malnourished. His height was 165 cm and his weight was 49 kg. His body mass index was 17.99. The abdomen was soft, with no tenderness or palpable masses. His history was significant for duodenal ulcers for more than 10 years. Laboratory tests showed a serum albumin level of 26.9 g/L, hemoglobin level of 12.8 g/dl, potassium level of 2.7 mmol/L, and sodium level of 139 mmol/L. Gastroduodenoscopy revealed a fistula with a diameter of 15 mm at the junction of the first and second parts of the duodenum, as well as an ulcer with a diameter of 8 mm next to the fistula . A biopsAfter discussion at the multidisciplinary team meeting, we concluded that the fistula was most likely caused by a chronic penetrating duodenal ulcer. After a detailed discussion with the patient, we decided to perform surgery. Severe protein malnutrition and electrolyte imbalance were treated with intravenous hyperalimentation.On March 17, 2022, surgical exploration indicated the presence of severe adhesions of the gastric antrum, duodenal bulb, and transverse colon, which were difficult to release. It is difficult to remove a fistula, and the fistula opening was located at the junction of the first and second portions of the duodenum. Accordingly, the patient underwent resection of the segmental duodenum, distal stomach, and partial transverse colon . Roux-enThe pathological examination showed chronic active inflammatory mucosa of the gastrointestinal tract with erosion and ulcer formation, stromal fibrosis, focal neutrophils, lymphocytes, and plasma cell infiltration. There was no evidence of tumor infiltration in the resected specimens .Postoperatively, it was necessary for the patient to fast. Anti-infection treatment, parenteral nutrition, albumin infusion, somatostatin, and gastric mucosal protective agents were administered. The patient was discharged from the hospital 9 days after surgery. An oral contrast study performed 3 weeks later revealed that the contrast media entered the gastric stump and passed smoothly through the anastomosis and into the jejunum with no obvious fistula . At the Duodenal ulcer is the most common cause of benign duodenocolic fistulas , 6. The The characteristic clinical features of a duodenocolic fistula include diarrhea, abdominal pain, and weight loss . Our patThe most reliable diagnostic tool is a barium enema, which has a sensitivity of 85% to 95% . Barium via endoscopy (via endoscopy is a potentially safer and less invasive approach. To sum up, fistula closure has been the most commonly performed procedure for the treatment of fistulas. Partial gastrectomy combined with gastrojejunostomy can be beneficial for severe adhesions and when dissection of the fistula is difficult (The fistula is a chronic disease, and the goal of treatment is permanent closure. Various treatments have been used to treat fistulas caused by benign diseases. We collected 15 articles involving 22 cases . Operatindoscopy . Therefoifficult , 23. Rigifficult , 23. A tifficult , 27. Herifficult .via endoscopy is a potentially safer and less invasive option. A case of benign duodenocolic fistula secondary to duodenum ulcer is presented. Surgery was performed. Postoperatively, his conditions improved dramatically, and he gained 11 kg. In addition, the etiology, clinical features, diagnosis, and treatment are reviewed.In conclusion, benign duodenocolic fistulas are rare and are associated with abdominal pain, diarrhea, and weight loss. The most reliable test for determining the presence of a duodenocolic fistula is a barium enema. Benign duodenocolic fistulas are usually treated by closure of the fistula. If obstructions or severe adhesions are present, then right hemicolectomy or partial gastrectomy may be treatment options. In some cases, the placement of an through-the-scope clip


We experienced a rare case of right-sided large bowl obstruction (LBO) of the colon caused by chronic diverticulitis, which was challenging to diagnose.A young male was admitted to our department with a fever, diarrhea, and right-sided lateral abdominal pain for several days. CT showed a thickened ascending colon wall with stenosis and adjacent retroperitoneal inflammation without marked diverticula. The next day, he developed severe abdominal pain, and perforation was suspected. We chose the “interval definitive surgery”; at that time, intestinal decompression and laparoscopic drainage. Colonoscopy showed an edematous membrane, but no cancerous lesions or diverticula. Hemi-colectomy was performed after 10 days' nutritional therapy. No postoperative complication occurred. The histopathology showed that the pathogenesis was chronic diverticulitis.There have been few reported cases of right-sided LBO caused by diverticulitis, but it is important to be aware that benign disease, such as chronic diverticulitis, can cause LBO. Initial conservative therapy and nutritional therapy produced a correct diagnosis and good outcomes.Performing “interval surgery” allowed us to make an accurate diagnosis and may help to prevent surgical complications in rare cases of right-sided LBO due to diverticulitis. •Most cases of large bowel obstruction (LBO) are caused by cancer.•Right-sided LBO of the colon due to chronic diverticulitis is rare.•Definitive interval surgery leads to better outcomes and reduces perioperative complications. He stated that he had not experienced any previous similar episodes and did not have an abnormal diet. He did not have any medical comorbidities and was not taking any medication. He smoked one pack of cigarettes per day and occasionally drank alcohol. A physical examination showed slight abdominal distention and right iliac hardness. His bowel sounds were diminished. A plain computed tomography (CT) scan demonstrated marked wall thickening in the ascending colon with intestinal distention and adjacent retroperitoneal inflammation, but did not show marked diverticulosis A and B. 2.1We had planned to perform colonoscopy after several days' fasting. However, the patient's abdominal pain worsened the next morning. A plain CT scan showed small air bubbles besides the ascending colon without fluid collection, which suggested micro-perforation of the distended intestine C. At thiFirst, a long transnasal tube was inserted for oral intestinal decompression. Then, preoperative colonoscopy was conducted, which showed stenosis of the ascending colon due to external compression. However, the colonoscope easily passed through the stenotic region. In addition, an edematous membrane was observed, but no obvious cancerous lesions or diverticula were noted A. ThirdlThe patient's peritonitis had completely resolved by postoperative day 4. A radiographic contrast enema performed through a long nasal tube showed the passage of fluid through the stenotic region, but severe stenosis was still observed . The pat2.2The stenotic region of the ascending colon had adhered to the abdominal wall , but no 3Most of the reported cases of LBO caused by diverticulitis were left-sided. In addition to our case, we found 6 other reported cases of right-sided LBO with diverticulitis  during aEscherichia coli O156:H7, Salmonella, and Campylobacter, are known to cause inflammation, especially in the ascending colon. Escherichia coli O156:H7 infections can cause ascending colon wall thickening of >20 mm on plain CT, but such thickening is generally accompanied by hematochezia and small amounts of ascites In the present case, infectious colitis was initially considered as a differential diagnosis due to the acute onset of the patient's symptoms in the absence of previous similar episodes. Some bacteria, such as Based on this case, we would like to highlight the importance of “interval surgery”. Our patient was diagnosed correctly and recovered steadily without any complications after definitive surgery because we had decided to perform minimal interventions; i.e., so-called “interval surgery”. In cases of complicated appendicitis, such as those involving perforated appendicitis, we usually choose interval appendectomy 4We reported a rare case of right-sided colon diverticulitis with LBO. Clinicians should recognize that diverticulitis is one of the causes of LBO. In addition, interval surgery supposed to be considered to achieve the best outcomes for such patients.None.N/A.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Kazuhiro Hiyama performed all clinical interventions and critically revised the final manuscript.Shogo Yoshida made substantial contributions to conception, design, data acquisition and interpretation of the study and was involved in initial drafting of the manuscript.Other authors were involved in revision of the final manuscript.All authors reviewed the final manuscript and give approval of the version to be published and agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.N/A.Kazuhiro Hiyama.Not commissioned, externally peer-reviewed.The authors declare that they have no conflicts of interest and that there are no relevant financial disclosures to report.


Infections associated with giant intradiploic cranial epidermoid cysts are rare. This case report describes the successful surgical management of a 71-year-old diabetic man with a giant intradiploic cranial epidermoid cyst associated with a secondary infection. The patient underwent successful resection of the infected lesion with washout, debridement, and obliteration of the eustachian canal and external auditory canal. At the six-month follow-up, the infection was resolved and the patient was doing well clinically. Intradiploic epidermoid cysts are rare, and the presence of a superimposed otogenic infection is exceptionally rare and infrequently reported in the neurosurgical literature. Cranial epidermoid cysts are encountered infrequently, accounting for 0.2% to 1% of intracranial tumors . AlthougA 71-year-old diabetic man presented with dizziness, low-grade fever, and chronic purulent otorrhea. He had a secondarily infected intradiploic mass arising from the occipital bone with erosion into the mastoid space and gross bony dehiscence of both the external auditory canal (EAC) and posterior fossa plate on computed tomography. Imaging showed occlusion of the left jugular vein and sigmoid and transverse sinuses, rim enhancement, diffusion-weighted imaging restriction, and significant mass effect upon the left cerebellum .Video 1: A left transtemporal-transoccipital approach for washout and near-total resection of an infected giant intradiploic epidermoid tumor.Used with permission from Barrow Neurological Institute, Phoenix, Arizona.The external auditory canal was over-closed, and all remaining epithelial elements, including the tympanic membrane and middle ear mucosa, were removed. The eustachian tube was obliterated, and the facial nerve was preserved throughout its course. Gross inspection revealed packed epithelial debris punctuated with microabscesses throughout. Infection transmission had occurred via erosion of the lesion through the bony EAC. Ultimately, after debulking, all visible squamous epithelial elements were stripped. Histopathology was consistent with epidermoid tumor and secondary inflammatory processes. To prevent a recurrence, the EAC was overclosed, and all remaining epithelial elements, including the tympanic membrane and middle ear mucosa, were removed. The eustachian tube was obliterated. The facial nerve was preserved throughout its course. Care was taken to keep the dissection extradural in regions of gross bony dehiscence, and a cerebrospinal fluid leak was avoided. After copious debridement and irrigation with bacitracin solution, the defect was coated with vancomycin powder. The cavity was obliterated in part with muscle turn-in flaps.Postoperative magnetic resonance imaging demonstrated heterogeneous tissue enhancing over the resection site and no infectious signs. At six months, the patient had near-complete tumor resection with no signs of infection Figure ; he had The patient gave informed consent for the case report, and the institutional review board at St. Joseph’s Hospital and Medical Center  approved the study protocol.Intradiploic epidermoid cysts are extremely rare and slow-growing brain tumors. These cysts commonly arise in the third or fourth decade of life as painless, well-circumscribed, subcutaneous lesions and are more common in men. Epidermoid cysts can result from abnormal embryonic development, or they can be secondary to trauma that traps epidermal tissue in the diploe of the skull. The location and cause of the tumor may lead to variable surgical complications and affect the optimal treatment modality for lesions, as in the case described above. The diagnosis of these lesions is usually suggested via imaging and confirmed through histological examination.Jaiswal and Mahapatra  reportedExtra attention should be given to preventing the infection of giant epidermoid cysts because of the likelihood of exposure to the external environment. Giant epidermoid cysts frequently occur within the frontal and temporal bones and can easily be exposed to pathogens through the external auditory meatus, orbit, or cribriform plate . Duan etBecause of their size and location, giant intradiploic epidermoid cysts often have extensive attachments to the surrounding bone and dura. Therefore, careful curettage is often necessary to fully liberate the tumor capsule from its dural attachments ,5. Most Recurrence of epidermoid and giant epidermoid cysts has been reported, usually in patients with incomplete resection . In addiComplete resection remains the treatment of choice for epidermoid cysts. Although patients can live for many years with these tumors, often asymptomatically, early surgery is most effective in preventing complications associated with tumor growth. This is a relatively safe procedure, even within a young patient population .Intradiploic epidermoid cysts are rare, and the presence of a superimposed otogenic infection is exceptionally rare and infrequently reported in the neurosurgical literature. We present a case involving epidermoid resection with infection down to the sternocleidomastoid and EAC. Each intradiploic epidermoid case is unique, and careful examination of the tumor’s relationship with the patient’s anatomy is essential before treating difficult lesions. Some complications may be unpredictable, and the surgeon must be meticulous in the analysis and approach for proper care.


There may not be a better time to talk about dysphonias then amidst the World Soccer Cup, when offices of otorhinolaryngologists are crowded with aphonic and dysphonic patients because of vocal-abuse laryngitis. Diagnosis in cases like these do not pose major difficulties, a careful interview coupled to a thorough and detailed physical exam are just enough.In this editorial we shall focus the most challenging topic in laryngology: the differential diagnosis of adduction spasmodic dysphonia, which includes vocal tremor and muscular tension dysphonia. Despite different etiologies, vocal symptoms are very similar in these cases, and the diagnosis can be very subjective, depending a great deal on the personal experience of the examiner.Adduction spasmodic dysphonia is a vocal disorder characterized by spasms of the laryngeal muscles during speech, causing broken, tense, forced and strangled voice patternsVocal tremor is characterized by a rhythmic laryngeal movement, causing rhythmic pitch and loudness alterations during speech. These laryngeal movements are often times followed by a tremor of the head and limbsTension dysphonia is associated to excessive muscular tension, both in the intrinsic and extrinsic laryngeal muscles, with an over adduction of the true or false vocal cords, and even sphincteric laryngeal constriction. This hyperfunctional pattern usually persists in various phonatory situationsThe differentiation among these three entities is not always easy for otorhinolaryngologists, since their main diagnostic tool, the laryngoscopic exam, does not show characteristic structural changes in any of the cases. To make matters even worse, there may be an occasional association between these conditions. For instance, patients with spasmodic dysphonia may present vocal tremor or muscle tension dysphonia associatedSince there are no clear morphological findings at the exam, many patients end up not being properly diagnosed and are eventually considered bearers of conversion or psychiatric disorders by family and physicians. Treated as such, often times they spend years without improvement. The psychological repercussion accruing from this situation may be catastrophic for the patient's social relations.Even for experienced laryngologists, used to dealing with disorders like this, diagnosis can be challenging. Incorrect diagnosis may lead to a less successful treatment. For instance, the use of Botulin toxin, treatment modality recommended for spasmodic dysphonia, for a patient with muscle tension dysphonia, may cause severe glottal incompetence, with severe hoarseness and aspiration of saliva and foodBecause of the challenging diagnosis of this condition, many investigators have studied methods that assess these patients in a more objective fashion. It is within this context that this month's issue brings the original paper “Adduction laryngeal dystonia: proposal and nasofibrolaryngoscopy protocol evaluation”. We still need other studies that may provide solid basis that allow us to carry out the differential diagnosis of spasmodic dysphonias in a more efficient way, and all studies in this sense are, of course, very much welcome.


It represents 10–15% of all malignant solid tumors in children.Acute urinary retention is an uncommon urinary symptom in young adults and it's mainly due to benign pathologies, such as urethral stricture. However, prostate RMS can be presented as voiding symptoms and urinary obstruction, so it must be included in the differential diagnosis.Early and multidisciplinary treatment of BP-RMS is mandatory. One of the main goals of treatment in children and young adults is to preserve the urinary bladder function to achieve a good quality of life in these patients. In an attempt to preserve these organs without affecting overall survival, a conservative approach based on polychemotherapy followed by radiotherapy or/and bladder-sparing surgery can be performed in some cases.52An 18-year-old healthy man attended to Emergency Department with acute urinary retention. He referred to a previous history of disuria without hematuria or other symptoms. Digital rectal examination showed and indurated mass, while laboratory blood and urinary test, as well as prostate specific antigen (PSA) levels were normal..Ultrasonography (US) and computed tomography (CT) scan showed a highly enlarged heterogeneous prostate of 61 × 65 × 74 mm (volume of 152 cc) with a poorly defined polypod heterogeneous mass with a maximum axis of 71mm .Picture Multiparametric magnetic resonance (MRI) revealed a heterogeneous enhancing mass replacing the prostate with infiltration to the right lateral wall of the bladder, ischiocavernosus muscles and intersphincteric space, respecting the internal and external anal sphincter. There were no lymph nodes involved .Picture The patient underwent transurethral resection of the prostate (TURP) and the histopathology showed fragments of an intensely necrotic neoplasm with histological characteristics of embryonal RMS . On the After pathological diagnosis, CT body was performed and no metastatic disease was found. According to a multidisciplinary committee's decision, the patient underwent induction chemotherapy based on 6 cycles of vincristine, actinomycin D and cyclophosphamide (VAC). Before VAC chemotherapy initiation, semen cryopreservation was performed. CT scan after the treatment showed a partial response so the patient maintained systematic treatment with chemotherapy adding local radiotherapy, followed by an intensification therapy with cyclophosphamide and vincristine (VC). However, in subsequents control CT and MRI there were no changes in the mass, so the patient finally underwent surgery, performing an uneventful radical cystoprostatectomy with ileal conduit as urinary diversion. Final specimen histopathology showed necrosis and fibrosis with absence of residual disease as well as free surgical margins. Finally, due to the aggressiveness of this type of tumor, the patient received adjuvant chemotherapy with 4 cycles of VAC and after a 2-year follow up he is currently free of disease.3BP-RMS treatment is a real challenge, especially in childhood and young adults. Multimodal treatment protocols  have been developed -over the years-by several cooperative groups to establish treatment strategies. An improvement in the 5-year overall survival rates for BP-RMS has been observed due to the application of these multimodal protocols, increasing estimated survival from 25% in the 1970s to greater than 70% nowadaysOne of the main goals in BP- RMS management in children and young adults is to preserve urinary function without compromising overall survival, so treatment must be carefully individualized according to the tumors histological features, location, extent and pathological stage.,Based on these premises, two main protocols can be highlighted: the European according to the International Society for Pediatric Urology (SIOP) and the American according to the Children's Oncology Group (COG) guidelines.In recent years, brachytherapy and proton beam therapy have been added to the multimodal treatment for local control strategy to reduce morbidity associated with radiotherapy.There are several options for urinary reconstruction after cystoprostatectomy. It is important to choose the best option according to the patient's age, tumors aggressiveness, location and extension and surgeon's experience. After radical surgery, the most frequently urinary reconstruction performed is an incontinent diversion with ileal conduit.Because BP RMS is an unusual and aggressive tumor in the young adult population, there are not enough studies assessing the long-term results regarding different urinary diversion types.4Although BP-RMS is an unusual tumor, it must be suspected in young adults with acute urinary retention. Early and multidisciplinary treatment is mandatory to achieve a good quality of life without compromising overall survival.The authors declare no conflict of interest.