Schwannomas of the trochlear nerve with the absence of systemic neurofibromatosis are considerably uncommon, especially complicated by intra-tumoral hemorrhage. Due to the lack of typical clinical manifestations and imaging findings, a definite diagnosis of trochlear schwannomas before surgery is particularly difficult.We report the case of a 64-year-old female patient who presented with a unilaterally intermittent headache of 2-month duration and without a remarkable neurological deficit at admission. Imaging studies revealed a well-demarcated cystic-solid lesion with mixed signals beside the brainstem and suprasellar cisterna. The patient underwent a surgical operation with total resection of the tumor by a subtemporal surgical approach. The tumor was intraoperatively found to originate from the trochlear nerve and was pathologically confirmed as a hemorrhagic schwannoma with cystic degeneration.We describe this case in detail and conduct a concomitant survey of the literature, summarizing the clinical presentations, radiological features, surgical treatment, and the possible mechanisms of hemorrhage in relevance to trochlear nerve schwannoma. In very2A 64-year-old female patient was admitted to our hospital due to a sudden onset headache on the right side 2 months ago with thereafter intermittent occurrence. The pain is with irregular patterns, occasionally accompanied by nausea. The patient had a 10-year history of hypertension and took antihypertensive drugs regularly with blood pressure controlled at a normal level. Unremarkable neurological deficits and no cutaneous signs of neurofibromatosis were found during the physical examination. Head computerized tomography (CT) scan, which was performed in the local hospital, showed an abnormal ovoid lesion with mixed density on the right side of the pons and suprasellar cisterna , headache , hemiparesis , ataxia , facial pain or paresis , and hearing loss n = 2; 4.6%). These extratrochlear symptoms may be explained by the tumor location, where the tumor commonly grows close to the tentorium and may compress the pons, the midbrain, and the adjacent cranial nerves  . As a re.6%. ThesRadiological imaging is the most utilized diagnostic technique in the evaluation of intracranial schwannomas. A typical schwannoma of the trochlear nerve on a head CT scan tends to appear as a single, well-demarcated, and solid lesion, located mainly in the course of the trochlear nerve and shows isointense or hypointense on T1-weighted, hyperintense on T2-weighted images, and intense enhancement after contrast administration at MRI . HoweverTrochlear nerve schwannomas were classified by Celli et al. mainly into three types based on the tumor location: cisternal, cisternocavernous, and cavernous types . CisternThe standardized guidelines for the management of trochlear nerve schwannomas have not yet been established . Howevervia spinal cerebrospinal fluid drainage, and then the temporal lobe was elevated after the division of several draining veins on the inferior surface of the brain tissue , lateral suboccipital (n = 10), transpetrosal (n = 7), and pterional (n = 5) approaches. The patients all have a good outcome with total  or at least subtotal  removal of the tumor. The selection of a certain approach is mainly based on the direction of tumor growth and the preference of the surgeon. In our patient, the classical subtemporal approach was used as in most prior cases. With this approach, adequate brain relaxation was achieved n tissue . The tumbbé vein .via retaining the tumor capsule or to repair the disrupted nerve, total resection of the tumor, if possible, is recommended by most scholars  of the patients during the postoperative follow-up. As the origin nerve of schwannomas, it is inseparable from the tumor intraoperatively. In addition, the trochlear nerve is the longest and thinnest among all cranial nerves, which made it prone to injury. Thus, the trochlear nerve would be hardly kept anatomically intact intraoperatively and tends to be sacrificed in the removal of the tumor . However of the pscholars , 45.Intratumoral hemorrhage is not uncommon for intracranial malignancies such as high-degree gliomas or metastatic tumors, and some benign tumors like large meningiomas. However, hemorrhage occurring in trochlear schwannomas is rarely encountered with only four cases reported to date. Of all four patients, acute onset of symptoms and signs was observed, including significantly increased intracranial pressure  , acute eThe pathogenesis of spontaneous hemorrhage from schwannoma is not fully understood. As with all tumors, rapid tumor expansion and inadequate blood supply may cause central necrosis, cystic degeneration, and consequent hemorrhage . The tro4Trochlear nerve schwannomas are rarely encountered worldwide with only a limited number of cases having been reported over the past decades. Due to the lack of specific symptoms and radiological features, a definite preoperative diagnosis remains a challenge. The therapeutic regimen includes clinical observation, stereotactic radiology, and surgical resection and needs to be individualized for each patient. Total resection can be achieved through various approaches, but trochlear nerve palsy is a common postoperative complication. Moreover, the potential mechanisms of intratumoral hemorrhage occurring in schwannomas may be multifactorial and need further research.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.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.All authors listed have made a substantial, direct, and intellectual contribution to the work and approved it for publication.


Lobular capillary hemangioma (LCH) is a benign, rapidly growing lesion affecting the skin and mucous membranes. LCH is common in the oral cavity but is rarely observed in the nasal cavity. The disease etiology is not fully understood; however, trauma and hormonal changes are attributable factors. Patients usually present with nasal obstruction and epistaxis. This study reports the clinical picture, diagnosis, and management of a rare case of bilateral septal nasal LCH. Lobular capillary hemangioma (LCH) is a benign, rapidly growing lesion that affects the skin and mucous membranes of the oral and nasal cavities . HoweverA 49-year-old female patient was referred from a secondary hospital for additional examinations and management. She had hypertension and hypothyroidism and presented with complaints of multiple episodes of bilateral epistaxis, nasal obstruction, postnasal drip, and facial pain. These symptoms persisted for nine months. She denied a history of allergies, recent infections, trauma, or chronic rhinosinusitis. She underwent right nasal mass excision and right-sided limited functional endoscopic sinus surgery (FESS) at the referring hospital. The biopsy results suggested LCH. Symptoms recurred a few months later, with reduced quality of life and depression. Clinical evaluation included nasal endoscopic examination, which revealed a bilateral reddish hemorrhagic polypoid lesion (approximately 1.0 × 1.0 cm) in the caudal region of the nasal septum bilaterally and opposing. The lesion occluded both nostrils and bled on touch Figure . No undeThe patient was prepared for endoscopic nasal mass excision under general anesthesia. Intraoperatively, a Killian incision was made caudal to the lesion on both sides. The mucoperichondrial flap was elevated until it reached the posterior, superior, and inferior margins of the lesion. There was iatrogenic septal cartilage perforation. Thus, the bilateral presentation in our patient may be due to the migration of a previous lesion through the perforation. The lesion was excised with the removal of the surrounding mucoperichondrium. To cover the bare cartilage area, an anteriorly based septal mucosal flap was harvested posterior to the lesion and rotated to cover the left side of the septum, where mucosal loss was relatively large. Silastic sheets were used.The postoperative course was uneventful, and the patient received oral antibiotics for 10 days, with a follow-up of 10 days. At follow-up, the silastic sheets were removed, and nasal endoscopic examination revealed bilateral caudal crusting adherent to the septum. Partial debridement was performed, and the nasal mucosa healed properly. The final diagnosis was LCH.Lobular capillary hemangiomas are benign lesions of the skin and mucous membranes. This type of hemangioma is also known as pyogenic granuloma, which is a misnomer because the lesions are neither contagious nor granulomatous. The lesions can be pedunculated or sessile and range from a few millimeters to several centimeters . LCH is LCH is a rare disease of unknown etiology. Pathogenesis may be related to hormonal, traumatic, viral, oncogenic, and other factors. LCH should be suspected when a vascular lesion is diagnosed in the nasal cavity. The treatment of choice is the surgical excision of the cuff of the surrounding mucoperichondrium. The bilateral presentation of LCH may be due to the migration of a lesion through an iatrogenic septal perforation in previously operated patients with residual lesions or recurrence. Although rare, LCH should be differentiated from a bleeding mass in the nasal cavity.


Benign nerve sheath tumours such as schwannomas commonly involve the peripheral and cranial nerves. A schwannoma in the adrenal gland is a very rare occurrence, which arises from the adrenal medulla. Its most common presentation is a non-functional incidentaloma. It does not have any unique imaging characteristic distinguishing it from other adrenal masses; hence, its diagnosis is usually confirmed by final histopathology. In this report, we present two cases of an adrenal schwannoma for which we anticipated an unusual diagnosis, which was confirmed through adrenalectomy on histopathology. Nerve sheath tumours such as schwannomas can arise from cranial and peripheral nerves of the head, neck, and extremities, retroperitoneum, or adrenal medulla ,2. They Clinically, most retroperitoneal schwannomas present with dull flank pain and abdominal discomfort or are detected incidentally during imaging for unrelated symptoms or at the time of autopsy ,5. The hCase 1A 44-year-old female with no comorbidities presented to our hospital with occasional left flank pain for three months. Initial ultrasound evaluation showed a left suprarenal mass of 6.5 × 6 cm with mixed echogenicity. Contrast enhanced-computerized tomography (CECT) of the abdomen was suggestive of a well-defined soft tissue lesion of size 6.5 × 5 × 6.5 cm in the left suprarenal region, with peripheral enhancement without any fat attenuation or calcification Figure . Fluorodeoxyglucose positron emission tomography (FDG PETCT) showed patchy tracer avidity in the left suprarenal region {Standard uptake value (SUV) max-13.5}, along with predominant necrotic areas with no evidence of any metastatic lesions elsewhere in the body Figure .Routine blood and hormonal workups, including serum cortisol, dehydroepiandrosterone sulfate (DHEAS), metanephrine, and normetanephrine, along with 24-hour urinary metanephrine and normetanephrine, were within normal limits, suggesting non-functional adrenal tumour. Laparoscopic adrenalectomy was performed, and intraoperatively, there was a well-encapsulated 6 × 6 cm suprarenal mass with multiple feeding vessels mainly from the medial side. Dissection was difficult, and intraoperatively, the tumour mass was felt to be hard and gritty when lifted using the suction device or grasper. The gross excised specimen on the cut section showed a homogenous solid component with yellowish nodules in between. Again, a gritty sensation was felt during cutting the specimen. The intraoperative and immediate post-operative phase was uneventful, and the patient was discharged on postoperative day three. The final histopathology revealed a well-encapsulated tumour composed of spindle cells with infrequent mitosis, areas of hyalinization, and cystic degeneration. Immunohistochemistry showed diffuse S-100 nuclear positivity pointing to a diagnosis of adrenal schwannoma Figure .. After two months of follow up the patient is symptomatically relieved with no evidence of any tumour recurrence or metastasis. Case 2A 45-year-old hypertensive female with a recent history of left flank pain on evaluation was found to have a large (11 × 10 × 7 cm) left suprarenal mass on the CECT abdomen  max-11.4 along with FDG-avid paraaortic and hilar lymph nodes. Blood levels of aldosterone, cortisol, DHEAS and urinary 24-hour metanephrine, and normetanephrine levels were within normal limits. After medical blood pressure optimization, the patient was taken up for left-open adrenalectomy and hilar lymphadenectomy. Intraoperatively, a stony hard left suprarenal mass of size 12 × 7 cm was found with lateral and inferior displacement of the ipsilateral kidney and maintained fat planes with adjacent organs. The suprarenal mass along with enlarged hilar lymph nodes was excised, and the specimen was sent for histopathological analysis Figure .The post-operative period was uneventful. The histopathology again revealed adrenal schwannoma with lymph nodes negative for metastasis.Schwannomas are benign nerve sheath tumours arising from cranial and peripheral nerves, with the VIII cranial nerve being the most common site. Most frequently found in the head and neck region, followed by the upper and lower extremities, trunk, gastrointestinal tract, liver, pancreas, kidney, brain, heart, and retroperitoneum . SchwannRetroperitoneal schwannomas are most commonly discovered incidentally in patients being evaluated for vague and non-specific abdominal pain. They are usually >4cm on diagnosis, as seen in our cases as well. Imaging through CT and MRI of a retroperitoneal mass localized to the adrenal region had differential diagnoses of adrenal adenoma, pheochromocytoma, myelolipoma, adrenocortical carcinoma, ganglioneuroma, and adrenal metastasis. Recently, based on the imaging characteristics through multi-detector computed tomography (MDCT), a well-defined unilateral mass with cystic degeneration septa and a characteristic progressive, contrast-enhanced pattern has been proposed as a unique hallmark of adrenal schwannoma, as seen in one of our cases .Functional evaluation through hormonal workup is always recommended for the management of any adrenal mass, including adrenal schwannoma. However, invariably the functional evaluation is negative in schwannomas. Both of our cases were suspicious of malignant adrenal neoplasm owing to the large size and imaging characteristics. PET CT is highly sensitive and specific for detecting adrenal malignancy along with metastases. We routinely obtained PET scans for all adrenal tumours >4 cm for which CT is not categorically suggestive of myelolipoma. PET CT helps rule out distant metastases. In both of our cases, PET scans showed high SUV max (>4) in the region of suprarenal mass suggestive of mitotic pathology.Surgical excision remains the gold standard treatment and diagnosis is established only by histopathology ,12. SurgA definitive diagnosis of schwannoma is obtained through the final histopathology with the tumour showing spindle cells having infrequent mitoses, along with areas of hyalinization and cystic degeneration with immunohistochemical S-100 antigen, vimentin, collagen IV, laminin positivity and keratin, desmin, CD 34 & CD117 negativity. Diffuse positivity for S-100 protein in the tumour cell cytoplasm is characteristic of tumours of neuroectodermal origin, along with unique nuclear freeze zones called the “Verocay bodies” .Malignant schwannomas are infrequent but are typically associated with Von Recklinghausen’s disease or Neurofibromatosis 1 (NF1) in 5-18% of the cases , with coAn adrenal schwannoma is a rare entity detected only after surgical excision of an adrenal incidentaloma and its histopathological analysis. Laparoscopic excision and open excision of the tumour are the most favourable procedures, with one being chosen based on feasibility. Non-functional nature of the tumour and the hard, gritty sensation during tumour handling and cutting of the specimen may be suggestive of an adrenal schwannoma; however, the definitive diagnosis is achieved through the final histopathological examination of the excised specimen.


Leiomyoma of the vulva is a rare, benign mass that is present on the vulva. Most commonly, they are described as painless, well-circumscribed, solitary tumors that are misdiagnosed as Bartholin cysts before surgery.A 45-year-old woman presented with a case of vulvar leiomyoma misdiagnosed as Bartholin cyst preoperatively. A solitary swelling mass measuring 3 cm × 2 cm was found in the left labia majora at the Bartholin gland site on physical examination.A vulvar mass extent and vascularity may be determined by imaging. A color doppler flow imaging of the posterior vaginal wall revealed abundant blood flow.To confirm vulvar leiomyoma, surgery and histopathology were performed.After 2 months of follow-up, there were no signs of recurrence in the patient.Rare vulvar leiomyomas are often mistaken for Bartholin’s cysts. It is also difficult to distinguish benign from malignant forms, making vulvar leiomyoma a difficult diagnosis. As there are a few techniques used to differentiate between the nature of the tumor, excisional biopsy seems to be the best current procedure employed in addition to being the treatment of choice for such tumors. Leiomyomas of the vulva are nonspecific and camouflaging. Vulvar leiomyomas are often painless, solitary, and well-circumscribed, however, they are sometimes preoperatively misinterpreted as Bartholin cysts, abscesses, or other benign disorders.,3Vulvar leiomyoma is a rare and benign mass on the vulva.We discuss the case of a 45-year-old woman who had a vulvar leiomyoma that was initially misdiagnosed as a Bartholin cyst.A 45-year-old female, gravida 1, para 1, resented to the clinic with a 1-year history of left labial mass. There was no history of discharge, fever, weight loss, or a history of malignancy in the family. General examination was unremarkable except for a soft mass that measured 3 × 2 cm in the left labial area  (Fig.  Vulvar Leiomyoma is rare. It comes from smooth muscle cells, spindle cells, and epithelioid cancer cells with eosinophilic cytoplasm, among other things. A vulvar myoma can appear at any age, and until the tumor is surgically removed and microscopic and immunohistochemical tests are performed, the condition is frequently misdiagnosed.A leiomyoma is a benign tumor caused by smooth muscle cells in any organ. In the current incident, a clinically verified Bartholin cyst was also present. Bartholin’s cyst and vulvar leiomyoma can be differentiated by the direction and consistency of the labia minora. If the labia minora are everted and the cyst is soft, it is a Bartholin cyst; if they are inverted and the cyst is hard, it is a vulvar leiomyoma. Vulval leiomyoma can also be detected in the clitoris or concealed by another condition.Vulvar leiomyoma is frequently mistaken as Bartholin’s gland cyst, the most common preoperative diagnosis, because the 2 conditions share some of the same presenting symptoms, such as a painless lump and swelling in the area. Excision by the surgeon is the treatment of choice.Using magnetic resonance imaging and transvaginal ultrasound, it is possible to diagnose vulvar leiomyoma and distinguish it from leiomyosarcoma. Regarding their view, our case lacked any of the aforementioned characteristics that suggested a benign leiomyoma diagnosis. Leiomyoma of the vulva should not be considered benign unless a histological study has been performed. Out of Nielsen et al 25 cases of vulval leiomyomas, 4 and 5 were found atypical or sarcomas, respectively. It is recommended, therefore, to surgically remove any vulval tumor for proper histological and immunological evaluation.Many vulval lesions look identical, making it difficult to tell benign from malignant lesions only by looking at them, so diagnosing smooth muscle tumors of the vulva can be difficult. A vulvar leiomyosarcoma may be suggested if 3 of the following criteria are met: the size of 5 cm or more, infiltrative margins, 5 or more mitotic figures per 10 high power fields, and grade 2 to 3 atypia.Leiomyoma of the vulvar is an uncommon tumor that is sometimes misinterpreted as Bartholin’s cyst. Leiomyoma of the vulvar is extremely difficult to diagnose since it is difficult to distinguish benign from malignant types. An unusual clinical presentation should prompt ultrasonography and/or magnetic resonance imaging to rule out malignancy, followed by surgical removal of the mass with a safety margin of normal tissue for histopathology and immunohistochemistry.The authors express their gratitude to the patient who made this work possible, as well as the professionals and researchers that participated in this study. A patient’s informed consent was acquired.Conceptualization: Jing He.Data curation: Jing He.Investigation: Wenhua Liu.Methodology: Wenhua Liu.Project administration: Wenhua Liu.Resources: Wenhua Liu.Software: Xiaoyu Wu.Supervision: Xiaoyu Wu, Dingheng Li.Validation: Dingheng Li.Visualization: Dingheng Li.Writing – original draft: Yuanwei Liu.Writing – review & editing: Yuanwei Liu.


Spontaneous or non‐traumatic rupture of the renal tract is an infrequent presentation, and it is most frequently caused by ureteric obstruction. Rupture could occur at any level of the upper urinary tract. However, it is most common at the renal calyces and complications that could arise include; urinoma, and or hematoma collection which could progress to abscess formation and sepsis. We report a 77‐year‐old male patient who attended the emergency department following referral from his general practitioner with a 6‐day history of progressively worsening left sided abdominal pain. Due to his co‐morbidities, presenting blood pressure and age, he was suspected of having an aortic dissection or ruptured abdominal aortic aneurysm and subsequently had a CT (computed tomography) Angiogram. This showed extravasation of contrast from the left kidney with a 12 mm obstructing vesico‐ureteric junction calculus necessitating urgent urology referral and prompt review. He was worked up for a ureteric double J stent insertion, however, the procedure was unsuccessful due to complex multiple urethral strictures. The patient subsequently had a nephrostomy inserted and was planned for optical urethrotomy, rigid cystoscopy, rigid/flexible ureteroscopy, and laser stone fragmentation of left obstructing vesico‐ureteric junction calculus. Spontaneous renal tract rupture is a rare presentation and commonly results from ureteric obstruction. It should be considered in suspected renal colic and management will, however, depend on the cause and level at which rupture occurs. The abdominal pain was of sudden onset and a constant ache scoring 8/10 on the numerical pain scale. His pain radiated to the back with no exacerbating or relieving factors and progressively got worse 3 days prior to presentation.The patient was an independent man with a WHO  performance status of zero. With regards to medical history, the patient was a known hypertensive and he had a previous pulmonary embolism. He suffered from chronic back pain from a work‐related injury and also had a suprapubic catheter due to multiple urethral strictures developed post green light prostatectomy. His chronic back pain was a mitigating factor toward his early presentation as he had attributed his chief complaint to an already existing pathology.General examination revealed an elderly man in no obvious distress. Cardiovascular examination revealed an elevated blood pressure . Abdominal examination revealed a suprapubic catheter in‐situ with no ooze or erythema around the entry port. Abdomen was soft and tender over the suprapubic region. There was no report of costovertebral angle tenderness. Bowel sounds were normal.3Full blood count, renal function tests, and serum electrolytes were within normal limits. He was investigated for aortic dissection with a computed tomography (CT) angiogram for suspicion of aortic dissection or ruptured abdominal aortic aneurysm. This revealed an incidental finding of intra‐abdominal free fluid on the left side related to a spontaneous rupture of the left renal calyces due to a 12 mm obstructing left vesico‐ureteric junction calculus Figures . The pat4The patient is a known hypertensive with previously documented abdominal aortic diameter reading of 55 mm. Coupled with his age, he was a prime candidate for possible aortic dissection.5Upon reviewing him at the emergency department, he was counseled on the findings from computed tomography (CT) angiogram. He was offered a rigid cystoscopy with left ureteric double J stent insertion to de‐obstruct the kidney and to return at a later date for a flexible/rigid ureteroscopy and laser stone fragmentation. He eventually had left nephrostomy on account of difficulty achieving urethral instrumentation due to multiple complex urethral strictures.There was no immediate post‐operative complication, and patient is awaiting optical urethrotomy, rigid cystoscopy, rigid/flexible ureteroscopy, and laser stone fragmentation of left obstructing vesico‐ureteric junction calculus.6Non‐traumatic renal tract rupture is an infrequently diagnosed urological condition, and outside of its scarce occurrence, a contributing factor to its rarity may be that the diagnostic investigation of choice, a computed tomography urogram, is not first‐line in the management of patients presenting with abdominal and flank pain. Patients presenting with flank pain are investigated with a non‐contrast computed tomography scan or an ultrasound scan if pregnant or children.A build‐up of intraluminal pressure within the renal tract is the mechanism that results in spontaneous rupture, and this increased pressure is often the result of ureteric obstruction, commonly by a stone. However, spontaneous rupture can also result in the setting of infections and tumors, which cause increased pressure in the renal pelvis.Patients with renal tract rupture present with a wide range of symptoms from mild flank pain that goes unreported to more serious symptoms of an acute abdomen resulting from the extravasation of urine. In two reported cases of renal rupture,Computed tomography urogram is the investigation of choice in the diagnosis of urinary collecting system leaks and urinomas. Diagnosis of spontaneous renal tract rupture is complicated by a low index of suspicion particularly in patients with several competing diagnoses. In this case, the patient, a known hypertensive with left sided abdominal pain, was suspected of having a ruptured abdominal aortic aneurysm. Consequently, he had a computed tomography angiogram where an incidental finding of renal tract rupture was made when contrast was seen extravasating into the peritoneal cavity from the lrenal pelvis Figures , 2 and 3The management of renal tract rupture is dependent on the level and cause of the rupture. A nephrectomy may be indicated in oncological causes. In other cases, however, immediate urinary de‐obstruction using a ureteric stent or nephrostomy allows the rupture to heal before definitive treatment which usually involves stone removal.7The patient attended for his planned optical urethrotomy, rigid cystoscopy, rigid/flexible ureteroscopy, and laser stone fragmentation 6 weeks after initial presentation. Urethroscopy was, however, unsuccessful due to an obliterated urethral meatus, and thus, cystoscopy was achieved via patient's suprapubic catheter site. Intraoperatively, the stone was not found in the bladder nor the left ureter or its orifice. The patient underwent another CTKUB  that showed the stone to be in the prostatic urethra, and he is currently awaiting cystolithotomy or cystolitholapxy.Mr. Emmanuel Chinedu Okpii took the patient's history, wrote and reviewed the manuscript. Ms. Fatima Adamu‐Biu and Dr. Kingsley Chibuikem Okpii wrote and reviewed the manuscript.The authors declare no conflict of interest.Ethics approval was not required; however, departmental permission was sought prior to publishing.The patient's written consent was obtained for the publication of this case report.


Penile strangulation is one of the rarest clinical findings. So, we aimed to present the clinical case of two cases that we encountered at our institute.Case 1. A 26-year-male with bipolar disorder presented to emergency room with complaints of metallic ring entrapment in the penis for 4 days. A gold-plated metallic ring measuring 1 × 0.25 cm was found encircling the coronal sulcus along with foul-smelling pus and slough underneath as a result of necrosis of the skin. Jumbo cutter was used to cut the ring. Loose sutures were applied to the injured part with debridement. Case 2. A 66-year-old man presented to emergency with complaints of penile swelling and poor urinary stream for the last 2 days. Local examination showed a plastic ring of water bottle seen stuck at the root of penis. Distal penis was edematous with multiple areas of skin discoloration and petechia. The ring was cut with simple surgical scissor.This clinically emergency condition may lead to wide range of vascular and mechanical injuries if the treatment is delayed. Plastic rings can be easily cut out whilst metallic objects which are thick and hard are difficult to remove. Pliers' application or use of jumbo cutter as in one of our cases is beneficial for prompt release of edematous pressure and salvage of the penis.Penile strangulation due to foreign body is a rare clinically emergency condition. Urgent intervention is necessary to remove the object causing constriction along the penis. •Penile strangulation due to foreign body entrapment is one of the rarer emergency conditions in urology.•This may lead to wide range of vascular and mechanical injuries if the treatment is delayed.•Urgent intervention is necessary to remove the object causing constriction along the penis. Mostly, this clinical condition is associated with an attempt of the individual for persistent arousal of penis for sexual gratification using the rings through the penis 22.1A 26 years male patient with the history of bipolar disorder under medication with mood stabilizers and anti-psychotics presented to emergency room with complaints of metallic ring entrapment in the penis for 4 days. The patient developed gradual progressive swelling of the glans of penis distal to the ring site. Following the incident, the patient tried to remove the ring, however remained unsuccessful. There was no history of intoxication or drug abuse. On local examination, a gold-plated metallic ring measuring 1 × 0.25 cm was found encircling the coronal sulcus. Gross edema and congestion of the glans penis and prepuce distal to the metal ring was seen along with foul-smelling pus and slough underneath the metal ring as a result of necrosis of the skin due to the strangulating effect A and B. 2.2A 66-year-old man, chronic alcoholic presented to emergency with complaints of penile swelling and poor urinary stream for the last 2 days. Patient gave history of insertion of plastic ring on the penis for the sexual gratification under the influence of alcohol. There was no history of trauma.During presentation, the signs of alcohol intoxication were not evident. Vitals were stable. Local examination showed a plastic ring of water bottle seen stuck at the root of penis. Distal penis was edematous with multiple areas of skin discoloration and petechia B. The ri3The cases of penile strangulation have been reported worldwide which is rare. Till date, around 60 cases have been reported in the literatures since Gauthier reported the first case in 1755 Bhat et al. in 1995 proposed a classification of penile incarceration which composed of 5 grades ranging from edema to necrosis as follows As per the classification, both of our cases fell upon Grade 2.Early intervention is deemed necessary to have the favorable outcome. Treatment depends upon the degree and material of entrapment along with distal edema associated with it. Nonmetallic objects like plastic rings can be easily cut out as one of our cases whilst metallic objects which are thick and hard are difficult to remove with chisel, saw or cutter 4Penile strangulation due to foreign body is a rare clinically emergency condition. Urgent intervention is necessary to remove the object causing constriction along the penis. Availability of pliers and removal tools should be assessed as soon as possible for quick removal and salvage of the penis.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.Ethical approval was not mandatory for publication of case reports as per the institutional policy.This case report did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Suman Baral.1.Name of the registry: NA2.Unique identifying number or registration ID: NA3.Hyperlink to your specific registration (must be publicly accessible and will be checked): NA.Design and Idea: Suman Baral, Ganesh Bhakta Acharya, Abishek PoudelDrafting: Suman Baral, Ganesh Bhakta Acharya, Abishek PoudelFinal Revision: Suman Baral, Ganesh Bhakta Acharya, Abishek Poudel, Ananda Neupane, Kamal Kadel.Authors declare that there are no any conflicts of interest regarding publication of the manuscript.


Solitary infantile myofibroma is a benign fibrous tumor occurring in early childhood. Although rare, it is the most common benign fibrous tumor of infancy. The clinical course of the disease is almost uniformly good since most tumors regress spontaneously. When indicated, conservative surgical excision is the treatment of choice, with a low recurrence rate. We present a case of solitary infantile myofibroma that recurred after three attempts of surgical excision, questioning the reported recurrence rate and the standard of care in recurrent solitary infantile myofibroma. Infantile myofibromatosis (IM) is a mesenchymal disorder characterized by myofibroblastic proliferation that results in solitary, multicentric, or generalized neoplasms affecting the skin, bone, and viscera . It was Although rare, IM is considered the most common benign fibrous tumor in infancy . It is tGain-of-function mutations of platelet-derived growth factor receptor-beta (PDGFRB) were found in affected children, especially those affected by the multicentric form of IM, albeit present in solitary IM . In addiA 10-year-old healthy boy presented to our dermatology department for a right cheek nodule. It appeared for the first time during early infancy. It was excised twice and recurred each time two to three months after its excision. The lesion was asymptomatic and didn’t generate any mass effect or physical limitation. The family history was unremarkable. On physical exam, two scars were identified on the right cheek and a firm, round, partially mobile, 3 cm subcutaneous nodule was palpated Figures .Based on history, clinical findings, and complete physical exam, the differential diagnosis included infantile fibrosarcoma, neurofibroma, dermoid, and desmoid tumors.Biopsy was taken and histopathology showed nodular proliferation in the reticular dermis composed of fusiform biphasic cells arranged in fascicles in a mixed  stroma without pleomorphism or atypia. These findings were consistent with a myofibroma Figures , 4.Computed tomography (CT) of the face showed that the myofibroma affected the skin and subcutaneous tissue without bony involvement Figures , 6.To rule out further tissue involvement, a CT scan of the chest, ultrasound of the abdomen, and X-ray of long bones were performed and came back negative.The tumor was excised based on the parents’ decision despite our advice for conservative management. Excision was complete with tumor-free margins on histopathology and the diagnosis of IM was re-confirmed based on the presence of fusiform biphasic cells arranged in fascicles in a mixed stroma. Unfortunately, the myofibroma recurred, once again, only a few months after its surgical removal.Infantile myofibromatosis/myofibroma is a rare fibrous disorder of childhood and infancy, its incidence ranges between 1 in 150,000-400,000 . IM is cClinically, IM presents as one or more rubbery to firm nodules. Lesions are usually asymptomatic, skin-colored to purple, and located in the dermis or subcutaneous tissue with the possibility of muscular, skeletal, and visceral involvement ,7. The hHistopathologically, IM is described as a well-circumscribed, non-capsulated mass characterized by a distinctive biphasic growth pattern. The center of the lesion is composed of immature, plump, myofibroblastic, spindled tumor cells with hemangiopericytoma-like branching blood vessels while the periphery consists of nodules and fascicles of hyalinized, myoid-appearing cells. Despite the impression of circumscription, IM sometimes demonstrates infiltrative growth. Note that myofibroma and myopericytoma share some features, suggesting that these entities could be related . ImmunohIn their review of the literature, Braun et al. found that 35% of published cases had solitary cutaneous or subcutaneous IM . Of thosFamilial cases have been reported without a clear inheritance pattern but our patient had no family history of IM . Gain-ofPatients may experience morbidity related to growth retardation, vocal cord paralysis, pathologic fractures, and physical limitations due to the mass effect . In vertAn initial increase in the number and size of the lesions was frequently reported, but the majority of IM cases (in the solitary as well as multicentric forms) follow the typical benign course with spontaneous regression in the first few years following the diagnosis. Different theories were postulated as explanations of this process but were never confirmed, in particular, the apoptotic mechanism and angiogenesis modulating factors ,5,6,10.Considering the frequent spontaneous regression, close observation remains the most appropriate approach in the majority of cases. When tumors affect vital functions or cause growth anomalies or cosmetic deformities, conservative surgical excision is the standard treatment ,13,14. IThere are no definitive guidelines for generalized IM treatment. Low-intensity chemotherapy with vinblastine-methotrexate, vincristine- actinomycin-D, or interferon-alfa or antiangiogenic therapy  might be considered in this form despite their side effects and unconfirmed efficacy ,6,15,16.The reported recurrence rate of IM after excision is 7-10% ,20. WhilInfantile myofibromatosis is a rare disease of infancy. When indicated, conservative surgery is the treatment of choice in solitary forms. Although IM seldom recurs, it recurred exceptionally after three attempts at surgical excision in our case, questioning the reported recurrence rate. Recurrent lesions could also be treated by chemotherapy with or without radiotherapy. Since recent literature supports targeted therapy by tyrosine kinase inhibitors, it could replace the standard aggressive treatments in primary IM as well as their recurrences. Further research is still required to assess their safety and efficacy.


Schwannomas are rare benign tumors that develop from Schwann cells that represent 0.3 to 0.4 cases per 100,000 persons per year. We report a case of pre-sacral schwannoma, a rare tumor, especially in the pelvic area. This case comes to help further teams in their management as its scarcity made any attempt to make proper recommendations obsolete.a 53-year-old otherwise healthy woman who presented with a 2-year history of right-sided sciatica. The radiologic characteristics of the mass suggested presacral schwannoma type III of the Klimo classification as the most possible diagnosis.Therefore, surgical resection was decided, and an anterior approach was chosen. By laparotomy a 6cm retroperitoneal encapsulated mass with no invasion of the adjacent organs. We performed a digital enucleation of the tumor through a capsulotomy.Pathology confirmed the diagnosis of schwannoma. There were no features of malignancy. The post-operative period was uneventful. A follow-up examination at 6 months showed no signs of numbness or weakness in the right leg. The previously described pain totally regressed.Although schwannoma is a benign lesion, it may become malignant, especially when associated with neurofibromatosis making its surgical removal primordial. Its pelvic location may make its diagnosis delayed due to non-specific symptoms mainly through compression of local organs. Its surgical management can be challenging due to large size tumors with adherence to peritoneal and retroperitoneal organs. Quality of the resection is important in the recurrence and necessity for reoperation. A multidisciplinary approach is therefore recommended to ensure optimal treatmentDue to its rareness, there is no clear consensus in on the management of schwannomas therefore we chose to write our case in order to further enrich the literature to achieve one-day guidelines for schwannoma treatment. •Schwannoma is rare.•Symptoms are nonspecific.•Surgery is the only permanent solution.•Surgical approach depends on shwannoma type.•Recurrence is possible in case of incomplete surgery. They are usually found in the neck and head region, but sporadic cases of pelvic schwannomas have been reported. They are usually asymptomatic until their growth compresses adjacent organs. Due to inconsistent symptoms, they are often misdiagnosed as pelvic lesions. We report a case of pre-sacral schwannoma, a rare tumor, especially in the pelvic area. This case comes to help further teams in their management as its scarcity made any attempt to make proper recommendations obsolete.This case report has been reported in line with the SCARE Criteria .2We report the case of a 53-year-old otherwise healthy woman who presented with a 2-year history of right-sided sciatica. The pain was greater in the supine position and slightly relieved when upright. There was no weakness or numbness. The symptoms began gradually 2 years ago with no history of trauma or injury.Initial management included an EMG that showed myelin damage to the right L5 root which indicates L5 root compression. MRI of the pelvis and lumbar-sacral spine revealed a well-defined ovoid mass in the right presacral space, adherent to the sacral anterior surface without any sign of invasion growth. The parenchyma was heterogeneous with few calcifications noted. The mass had contact with the external iliac vein, internal iliac artery and vein, and the right ureter . The radTherefore, surgical resection was decided, and an anterior approach was chosen. We found it necessary to put a double J stent in the right ureter prior to the operation to prevent its injury during the pelvic dissection.The patient was placed in a supine position and a lower midline incision from the umbilicus to symphysis pubis was performed. After opening the abdomen, a 6cm retroperitoneal mass was detected occupying the right presacral space and compressing the vascular structures. The ureter was identified and protected. The vascular structures were thoroughly dissected around the lesion. The mass was encapsulated with no invasion of the adjacent organs . We perfPathology revealed a well-encapsulated tumor of interwoven spindle cell bundles. There were areas of calcification within the tumor. These features were typical of a degenerative schwannoma, and this was confirmed on immunohistochemistry with component cells staining strongly for S-100 protein. There were no features of malignancy .Fig. 3ScThe post-operative period was uneventful. A follow-up examination at 6 months showed no signs of numbness or weakness in the right leg. The previously described pain totally regressed.3Schwannoma is a rare nerve sheath tumor (NST), as it represents 0.3 to 0.4 cases per 100,000 persons per year, with most cases presenting around the fifth decade, which is similar to our patient ,3.What is interesting in our case is also the pelvic nature of the mass. Indeed, sacral localization of this tumor is quite rare as it represents less than 3% of all schwannomas .This unique location and the fact that there is plenty of space to develop in is why presacral schwannoma is quite an insidious tumor. As it is pointed out in our case the patient began experiencing right radicular pain 2 years prior to the final diagnosis. This non-specific and very common symptom was treated as such with antalgics and non-steroidal anti-inflammatory. This is concurring with the literature, as some authors suggested that the onset of clinical manifestations ranges from 1 to 7 years according to literature with an extreme of 25 years in one case .Now this delay in the diagnosis may be acceptable since schwannomas are mostly benign but bear in mind that 5–18% of them are associated with neurofibromatosis, which may become malignant .The rareness of the disease and its pelvic location made it challenging for us to find conclusive recommendations to help with our management, but we did find Klimo et al. proposition for the classification of sacral NSTs interesting. Klimo et Al. suggested a classification scheme based on their location regarding the sacrum and were so divided into three types. Type III, as depicted in the classification, is primarily located in the presacral/retroperitoneal region similarly presented in our case . Now thiThis classification helped us in preoperative planning, involving a multidisciplinary approach with a neurosurgeon, an orthopedist, and a general surgeon. We did not need the intervention of a urologist in operative, but we did find it useful to put a double J stent in the right ureter prior to the operation to prevent its injury during the pelvic dissection.What we find interesting in our case is that although authors suggested that sacrificing nerve roots carries a minimal risk of debilitating postoperative neurologic deficit , we chosNow we are aware that this approach may expose to the risk of recurrence as it may be considered as a subtotal resection. This was noted in Conti P et l. Analysis that concluded to a 4.5% recurrence , all of However, we do plan on conducting an annual surveillance regimen for our patient by clinical assessment and an MRI to detect an eventual local recurrence at an early stage of its evolution making reoperation easier.4Although schwannoma is a benign lesion, it may become malignant, especially when associated with neurofibromatosis making its surgical removal primordial. Its pelvic location may make its diagnosis delayed due to non-specific symptoms mainly through compression of local organs. Its surgical management can be challenging due to large size tumors with adherence to peritoneal and retroperitoneal organs. Quality of the resection is important in the recurrence and necessity for reoperation. A multidisciplinary approach is therefore recommended to ensure optimal treatment.Not applicable.No sources of funding.Magherbi Houcine, CONCEPTUALISATION, REDACTION, DATA CURATION, PROJECT ADMINISTRATION.Ouadi Yacine, CONCEPTUALISATION, REDACTION, DATA CURATION, PROJECT ADMINISTRATION.Hammami Mahdi CONCEPTUALISATION, REDACTION.Zehani Alia PHOTOGRAPHY RENDERING.Boukriba Seif PHOTOGRAPHY RENDERING, DATA CURATION.Zaraa Mourad DATA CURATION.Fterich Fadhel Samir SUPERVISION, VALIDATION, VISUALISATION.Montasser Kacem SUPERVISION, VALIDATION, VISUALISATION.1.Name of the registry:2.Unique Identifying number or registration ID:3.Hyperlink to your specific registration (must be publicly accessible and will be checked):Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.OUADI YACINE.Not commissioned, externally pee-reviewed.All authors declare that they have no any conflicts of interest.


The urachus is an embryologic remnant of the cloaca that usually degenerates after birth, resulting from the obliteration of the allantois, whose role is to connect the bladder to the umbilicus. Incomplete removal of the lumen may give rise to different malformations of the median umbilical ligament after birth. Although in the pediatric population urachus are common, most cases are asymptomatic and may go unrecognized until adulthood and give rise to cysts, rarely reported in the literature. Thus, in this manuscript we present the circumstances of a 43-year-old Romanian woman showing hypogastric pain of moderate intensity for three weeks, radiation in the left lower limb, menstrual cycle abnormalities, and dysmenorrhea. Based on the initial examinations, a paraovarian cyst measuring 80 mm was noted. Through the subsequent magnetic resonance imaging (MRI) conducted, a hypoechoic mass was detected, and the patient underwent a tumorectomy and partial cystectomy. A 9.7/7.5-cm tumor was excised, and the anatomopathological result was urachal mucinous cystadenoma. It came to our attention that relatively scarce data were found in the literature, with only seven studies with the diagnosis of the urachal cyst. The urachus is a three-layered allantois-derived embryologic remnant that develops before the fifth month of fetal development. Taking the shape of a fibromuscular tubular structure that descends into the pelvis, created through lumen obliteration, it degenerates after birth .The primary role is to connect the dome apex with the umbilicus, compartmentalized by umbilico-vesical fascia along the medial umbilical ligament . PostnatHowever, there are occasions when this mechanism fails, which is not an uncommon phenomenon, and the epithelialized duct may persist until adulthood. The first documented primary urachal cancer (UC) was described in 1863 by Hue and Jacquin, responsible for around 0.5% and 20–40% of bladder malignancies and adenocarcinomas [Thus, urachal anomalies (UA) caused by partial or incomplete obliteration could give rise to five distinct malformations. According to the latest figures, among the common UA forms stand congenital patent urachus 47%), urachal cyst (30%), umbilical-urachal sinus 18%), vesicourachal diverticulum (3%), and alternating sinus [%, uracha%, vesicoAlthough the urachal cysts incidence is one in 5000–150,000 in the adult and pediatric population ,12, withUnfortunately, due to infections and malignancy processes, if they do not resolve in the first two years, this might be the transition phase for the patients to become surgical candidates. In the absence of complications, anomalies are found incidentally for other medical reasons ,15.On the other hand, 35% of patients may present to the Emergency Department reporting lower abdominal pain , signs oThus, this manuscript presents the case of a Romanian female patient with a urachal mucinous cystadenoma initially misdiagnosed with a paraovarian cyst.A 43-year-old Romanian female patient (M.P.) presented to the “Cuza Voda” Obstetrics and Gynecology Clinical Hospital from Iasi, reporting hypogastric pain of moderate intensity whose onset started three weeks prior, with radiation in the left lower limb and accompanied by menstrual cycle abnormalities and dysmenorrhea.She declared having irregular scanty flow during menstruation, and dysmenorrhea, with two previous Caesarean (C)-sections. Moreover, she suffered a cholecystectomy complicated by a septic condition, but her bladder and bowel habits were normal.During the standard evaluation, an enlarged abdomen in volume was observed due to the adipose panicle, sensitive to palpation in the hypogastrium and Pfannestiel-associated scars following the C-sections and cholecystectomy. She did not complain of urinary symptoms, such as dysuria, pollakiuria, hematuria, or micturition disorders.Furthermore, the laboratory test results indicated anemia, with the value of the tumoral antigen CA125 of 132.3 U/mL (0–35 U/mL) and human epididymis secretory protein (HE) of 28.9 pmol/mL (<70 pmol/mL), indicating a risk of ovarian malignancy algorithm (ROMA) of 7.4% (<7.4%), also with the presence of calcium oxalate crystals in the urine exam.Considering all the information gathered, we first decided to perform an ultrasound (US) which revealed a large hypoechoic cystic formation on the left of the uterus, misdiagnosed as a paraovarian cyst measuring 80 mm .The subsequent MRI revealed slight motion artifacts due to intestinal peristalsis.The bladder had homogeneous content, without intraluminal protruding formations; imprinting of the medial and left-sided bladder dome by the dominant left adnexal lesion.The uterus located paramedian right was enlarged, with global dimensions of approximately 145 mm longitudinal/75 mm sagittal/90 mm transverse, and the endometrium had ~6 mm thick with no particularities. There was a diffuse thickening of the junctional area of the anterior uterine wall, up to ~42 mm, suggestive of adenomyosis. The junctional site thickness of the posterior wall was within normal limits (up to ~8 mm). The elongated cervix, up to ~45 mm, did not present macroscopic suspicious tumor lesions evident on magnetic resonance (MR).The right ureter was slightly ectatized posteriorly and cranially by the uterine fundal region (diameter 4–7 mm), probably by a degree of compression by the enlarged uterus. The upper pole of the uterus was located approximately next to the lower vertebral plateau L4.The uterus came posterior to the right ureter, the right common iliac vascular bundle, and its bifurcation. Anteriorly, it came into contact with the anterior pelvic wall , laterally with the right external iliac vascular bundle, medially with the described cyst, infero-medially with the urinary bladder, and cranially with intestinal loops.The right ovary, with a long axis of about 30 mm, was located right parauterine, ascending in the right iliac fossa, immediately caudal to the ileo-cecal region, showing some infracentimetric follicular images.The left ovary, with the long axis of about 32 mm, was located between the anterior pelvic wall and the left external iliac vascular bundle, about 7 cm lateral to the midline showing some follicular (possibly cystic) images up to ~16 mm.Left parauterine, between the left lateruterine wall and the left ovary, was attached to the anterior contour of the uterine round ligament, and an ovoid cystic lesion of approximately 100/68/50 mm (oblique CC/AP/LL) is evident .This showed probably proteinaceous fluid content and mildly irregular wall thickness up to ~4–5 mm  and with a mural micronodule of ~5 mm with contrast uptake at the level of the left antero-lateral contour. The appearance advocated the first hypothesis for a “border-line” left paraovarian cyst, with suspicious elements of neoplastic transformation . It presThe rectal ampulla did not show suspicious tumor masses. Several infracentimetric pelvic lymph nodes, without specific character, were noted, as well as a minimal plate of intrapelvic fluid with a millimeter thickness. No notable collections in the pelvic recesses. No suspicious focal bone lesions were evident in the pelvis on the acquired images. Diastasis of the rectus abdominis muscles in the pelvic region (distance of about 30–35 mm) could be observed .We decided to recommend a laparoscopy, but unfortunately, the patient refused this approach accusing the unfavorable evolution of a previous laparoscopic cholecystectomy. Thus, we performed a surgical intervention via laparotomy, and a tumor formation of 9 cm adherent to the anterior abdominal wall was identified . After tMacroscopic examination: (1) There was a tumor formation of 9.7/7.5 cm, with a small lack of substance on the cystic section, unilocular, with gelatinous, viscous content, thin wall, without internal vegetations. (2) Bladder wall fragment was 4/1/0.8 cm.Microscopic examination: (1) Cyst walls represented by conjunctive-muscular tissue and adherent adipose tissue, wallpapered only focally on a slope of cylindrical unistratified epithelium with apical mucosecretion, without atypia, next to extended beaches of mucus in which multinucleated giant cells and inflammatory cells were observed, floating epithelial cells. Intramurally, focal calcifications, and mucus focally dissecting the cyst wall were noted. (2) Bladder wall fragments represented by connective-muscular tissue, with notable congestion and chronic inflammation were evident.The urachus, known as the median umbilical ligament, is a midline fibrous remnant structure of the cloaca via the obliteration of the allantois that extends cranially to the umbilicus and degenerates after birth, laying between the peritoneum and the transversalis fascia , the priEscherichia coli, Enterococcus faecium, Klebsiella pneumonia, Proteus, Streptococcus viridans, and Fusobacterium [Thus, the cysts are vestigial remnants that can lead to complications, particularly affecting the males, and can be asymptomatic until adulthood upon becoming infected, including bladder fistula formation, cyst rupture, peritonitis, and sepsis, caused by acterium ,21. Besiacterium ,23,24.There are publications concerning urachal cysts, but those referring to malformations of 7 cm are relatively limited. Therefore, we found it appropriate to summarize the main aspects that are tangent with our case report in n = 176 patients aiming to associate the clinicopathological findings with malignancy in adults through univariate and multivariate logistic regression models. They identified urachal remnants in n = 46 children and n = 130 adults, from which n = 46, 43% (n = 20) presented with umbilical drainage, and 50% (n = 23) were diagnosed by physical examination and n = 34 children, 74% underwent a simple excision. Concerning the adults, 50% (n = 65) exhibited hematuria, 60% (n = 78) needed cystoscopy, and 41% (n = 53) CT, but 51% (n = 66) involved comprehensive operation, such as partial or radical cystectomy. Cumulatively, 51% (n = 66) were malignant, older patients (≥55-years-old) suffering from hematuria classified as strong predictors (p < 0.001), once with aging.Ashley et al.  conducten = 16 children diagnosed with urachal anomalies having abdominal or urinary symptoms, n = 8 being subjected to an open excision (5.5 years average—4 months–13 years-old), and n = 8 by laparoscopic surgery (10 years average—1 month–18 years-old). In this manner, the authors showed a mean operative time of 63 min (35–105 min) and 50 min (35–90 min) in both groups with no postoperative complications, discharge interval between 48 and 96 h, and a benign urachal remnant in each case.Subsequently, Chiarenza and Bleve  performeConsidering all the aspects mentioned above, we performed further searches in the literature and even fewer entries were returned with the result of urachal mucinous cystadenoma depending on the age of the patient, size, and histology.The diagnostic criteria panel has undergone revisions throughout the years, and the accepted ones are those according to Hamilou et al.  and descDespite the discrepancies in the current literature concerning this matter, there is pinpointed an increased interest in the prophylactic surgical removal of asymptomatic urachal remnants. Even though experts in the field advocate surgical interventions in both pediatric and adult groups whether they are (a)symptomatic, there are also situations when patients with non-specific atretic urachal remnant and specific symptomatic patients are be managed non-operatively ,37. In aSome authors support the removal of non-infected urachal remnants as an alternative to avoid future emergency surgery because it carries a higher risk such as infection and malignancy . In contContinuing with this concept, some advocate for a two-stage management technique involving the usage of antibiotics and US-guided drainage prior to the operative excision for infections ,40,41, wNevertheless, it should be emphasized that most asymptomatic cases in earlier stages of development may further reflect in diagnostic delays and poor prognosis, with it even being theorized that a neglected situation might become silent and culminate in infections, chronic inflammation with an increased risk for carcinogenesis , recurreImaging techniques, such as computed tomography, MRI, and ultrasonography (USG), constitute the radiologic core method of choice and play a pivotal role in differentiating the urachal cysts from other causes ,42, provThe peculiarity of the case lies in the rarity of the pathology, although it is not malignant. To the authors’ best knowledge, this is the only one reported in our country and in the northeastern region of Europe. The pathology is still difficult to diagnose preoperatively. A non-concordant imaging diagnosis, as in the presented case, can lead to incorrect management and affect the prognosis, especially in monodisciplinary hospitals, with limited resources, requiring a multidisciplinary team. The case draws attention to this pathology and the importance of a correct imaging diagnosis, especially in areas with limited resources, in order to direct the patient to a specialized center.


Fetus in fetu (FIF) is a rare congenital anomaly of asymmetric monozygotic twins, where the parasitic twin develops abnormally inside the body of the host twin. In most cases, it is incorporated into the sibling’s abdomen, which frequently presents as a retroperitoneal mass. Currently, at least 200 cases have been reported worldwide, being this the first case in Nicaragua. We describe a case of a male newborn, born via cesarean section, with a history of multiple congenital malformations observed via ultrasound examination. At birth, a mass is observed on its dorsum that impresses a skull, but without the presence of bones, with three limbs, two upper and one lower, with an outline located transversely on the pelvic girdle and the presence of two male genitalia with agenesis of the testicles and an accessory kidney. A preoperative diagnosis of FIF and spinal dysraphism was made by computed tomography (CT) and magnetic resonance imaging (MRI). They shared a spinal cord and had the presence of an open spinal defect type meningocele with aberrant roots. After the diagnosis and discussion, the multidisciplinary team proceeded to surgery to perform the separation of the twin (FIF). The subsequent anatomopathological examination revealed that the fetus was anencephalic and had reliable FIF characteristics. The resection was performed followed by the closure of the 430 mL meningocele and complete separation of the spine and the parasitic twin. We present the first case of fetus in fetu in Nicaragua. Fetus in fetu (FIF)  is a rarThere is a male predominance, and most are reported before two years of age . TreatmeWe present a male newborn of 36 + 6 weeks of gestation with a prenatal ultrasound report of multiple malformations. Physical examination showed multiple malformations Figure -1F. UltrComputed tomography (CT) angiogram of the thorax, abdomen, and pelvis reported dextrocardia, lumbar spine morphological changes at the level of the posterior elements from L2 to the most caudal portion, a sacral bone vestige, and lack of fusion of the vertebral body of L1-L2. Three iliac bones are identified, which articulate with the sacral vestige and with three femoral bones. The left lower limb is poorly formed. There is an incomplete bony pelvis where the parasitic limb articulates with dislocation of the same, which presents normal morphology.Adhered posteriorly adjacent to the retroperitoneum is a hypodense, oval, well-defined image of a parasitic fetus, with attenuation values of 3 and -6 Hounsfield units (HU), which could correspond to a second abdominal cavity occupied by liquid fat. This image measures 97 × 83 × 93 mm, and inside, there is an image of reniform morphology measuring 35 × 24 × 30 mm, which can be related to rudimentary kidney that shows uptake of contrast medium.On the 26th day of postnatal life, the multidisciplinary team composed of a pediatric neurosurgery, anesthesiologist, plastic surgery, orthopedics, and pediatric surgery performed a surgical intervention with the following findings: pelvic limbs, sacral remnant and atrophic hand without bone tissue, cutaneous appendix with two pedicles as a bridge that were disarticulated by orthopedics, and resections of mass of approximately 6 × 4 cm in size corresponding with supernumerary kidney, with artery, accessory renal vein, and penis with supernumerary scrotum. By pediatric surgery, it was found that it was not communicating with the healthy twin. We proceeded to perform a midline exposure from T12 to L2 through the spinal defect (bifidus), finding the absence of posterior elements, nerve roots that went to the defect sac (meningocele) containing 430 mL of yellow cerebrospinal fluid (CSF) with lumps, and dural defect of approximately 2 cm with nonfunctional roots inside; no nerve placode was found.We proceeded to resect the meningocele, rudimentary dysfunctional roots attached to the sac, and excess dura mater. The dura was closed with 5-0 Prolene blunt sutures, thus isolating the spinal cord and spinal defect of the healthy twin. Valsalva maneuvers were performed to verify the absence of CSF fistula. The muscle and lumbosacral fascia were closed with blunt tip 4-0 Vicryl.Then we proceeded to resect the remaining vestiges of the skin and muscular structures of the parasitic twin Figure -2D. PlasHe was extubated from the operating room to neonatal intensive care unit, and his evolution was satisfactory. He was discharged 26 days after surgery.The term “fetus in fetu” was first used by Meckel during the late 18th century as a rare condition where a malformed parasitic twin resides in the body of its host , with anFIFs are usually located retroperitoneally along the ventral midline , while oImaging tests play a key role in the diagnosis of FIF . UltrasoOnce FIF is diagnosed, surgical treatment is essential, and the prognosis is good following complete resection . FIF is LimitationsAlthough the various components of FIF are mature, there is still a possibility of malignant transformation. Sewell et al. reported that malignant transformation occurred after FIF resection . TherefoAlthough FIF is a congenital malformation that is rarely encountered in clinical practice, it is potentially harmful to the human body, especially when the patient is a newborn. The development of prenatal diagnostic techniques and the popularization of imaging technology have increased the rate of diagnosis of FIF. The multidisciplinary approach and intervention of the pediatric neurosurgery specialty were of great importance since intervening on the associated neural tube defect decreased neuroinfectious complications, improving neurodevelopment and the quality of life of the child and family.


Many conditions are known to cause chronic wrist pain, one of which is accessory muscles which can be easily overlooked as the cause of pain. Here we present a case of chronic wrist pain in a 33-year-old male who presented with painful dorsal unilateral right-hand swelling associated with increased activity. This patient was referred to the radiology department and was found to have an accessory muscle along the dorsal aspect of the wrist consistent with the extensor digitorum brevis manus muscle. The report includes the clinical presentation, radiologic findings, and management.  A variety of accessory muscle variations in the hands had been described throughout the literature. These accessory muscles are discovered incidentally during surgeries, postpartum autopsy, or by using advanced imaging techniques -2. TheseIn the prenatal phase, the first indication of upper limb musculature is observed in the 7th week of development as condensation of the mesenchyme derived from dorsolateral cells of the somites . The preClinically, EDBM is one of the causes of what has been described as fourth compartment syndrome which is chronic dorsal wrist pain in the fourth compartment of the hand . TherefoA 33-year-old male presented to the plastic surgery clinic complaining about a swelling in the dorsal aspect of his right hand that only became painful three months prior to his presentation. The swelling was noticed since his childhood and progressively increased in size. The pain was increasing with activities and was relieved with rest. Furthermore, the patient reported that the swelling resolves sometimes therefore our impression was Ganglion cyst and we planned for aspiration. However, the patient also reported that the swelling splits into two discrete swellings therefore we opted for magnetic resonance imaging (MRI) imaging. The patient did not report any other symptom such as numbness, weakness, or swelling on the contralateral hand. The patient did not have any past medical or surgical history. On examination, there was discrete swelling on the dorsal aspect of the right hand that became more prominent with dorsiflexion of the right hand and fingers Figure . No discThe patient was then referred to the radiology department for MRI. MRI of the right hand was obtained with gadolinium administration. There is an accessory muscle between the 2nd and 3rd extensor compartments of the dorsum of the wrist, consistent with the extensor digitorum brevis manus muscle. The ligaments, carpal bones, and joints were unremarkable  originates from the extensor surface of the hand/wrist, the most common origin described is either the dorsal metacarpal surface, the distal end of the radius, or the proximal portion of the radiocarpal ligament -10. The The action of EDBM was examined in most literature, which if stimulated deviates the proximal phalanx to the side on which it is inserted in relation to the extensor digitorum communis . Gama C EDBM can be diagnosed by electromyography or by MRI like in our case. If the diagnosis was still in question, surgical exploration can be utilized . The usual presentation of EDBM is a painless lump on the dorsum of the hand, however, many patients present with pain related to EDBM associated with repetitive use typically on flexion. Pain is explained by the constriction of the muscle belly by the extensor retinaculum, also termed the fourth compartment -12.The recommended treatment in most literature was conservative unless the patient presented with pain. If a patient presents with pain, the initial management is with rest, heat, ice, bracing, and/or therapeutic exercise . Wendel In conclusion, we hereby present a case of EDBM that presented as a painful palpable mass on the dorsum of the hand. The diagnosis of EDBM can be misleading and easily overlooked, and can be confused with a ganglion cyst. Therefore, proper history taking and examination skills and choosing proper diagnostic modality such as MRI imaging are all necessary to properly diagnose EDBM.


Superficial siderosis of the central nervous system is a chronic condition characterized by hemosiderin deposition in the brain and spinal cord. It's diagnosed by brain MRI. It can be caused by low-grade extravasation of blood into the subarachnoid space of the brain. There are 2 types of superficial siderosis cortical and infratentorial. Although asymptomatic in many cases; Cerebellar-predominant siderosis, a subtype of infratentorial, can affect hearing, gait, and even muscles. In this report, we present a case of a 51-year old female with complaints of hearing loss, unsteadiness in his lower limb, and spastic paresis. During MRI neuroimaging, we noticed findings of hypointensity areas within the brainstem and cerebellum, probably due to hemosiderin deposition. Based on the MRI findings, the patient was diagnosed with superficial siderosis. The patient was started on deferiprone and followed for the consecutive 18 months. Moderate improvement of the hearing loss and ataxia was noted while no change in muscle force. However, the repetitive MRI did not reveal any changes compared to the previous one. Superficial siderosis (SS) of the central nervous system (CNS) is a chronic condition characterized by hemosiderin deposition in the brain  subpial layers as a result of chronic or intermittent low-grade extravasation of blood into the subarachnoid space Cortical SS—Superficial hemorrhage in the cortical sulci causes hemosiderin deposition on localized supratentorial cerebral convexities ,4. It isInfratentorial SS—Brainstem and cerebellar-predominant SS, also known as infratentorial SS, Hemosiderin deposition occurs in the posterior fossa, brain stem, and spinal cord Superficial siderosis leads to progressive and irreversible neurological dysfunction A 51 years of age patient arrives at our clinic with complaints of hearing loss, unsteadiness in his lower limb, and spastic paresis. The complaints started 14 years prior with slow progression over the consecutive years. General physical examination revealed no significant changes. Further investigations for a history of trauma, surgery, and tumors were absent. No delayed recall score was present on mental examination. On neurological examination, sensorineural hearing loss, a weak finger to nose test, and reduced force of the lower limbs. Laboratory tests were within range, including blood and urine analyses, blood coagulation, liver and kidney functions, thyroid tests, and vitamin concentrations. In addition, the patient's antinuclear antibody, anti‑O antibodies, erythrocyte sedimentation rate, and tumor markers were all normal. During MRI neuroimaging, we noticed findings of hypointensity areas within the brainstem and cerebellum, probably due to hemosiderin deposition. Based on the MRI findings, the patient is diagnosed with Superficial Siderosis. The patient is started on deferiprone and followed for the consecutive 18 months. Moderate improvement of the hearing loss and ataxia, while no change in muscle force. However, the repetitive MRI did not reveal any changes compared to the previous one.Superficial siderosis consists of hemosiderin deposition within the CNS inner layers . It can be associated with any form of CNS hemorrhage including SAH, SDH, and primary intraparenchymal/intraventricular hemorrhage [2]The pathologic changes associated with SS have been previously described. Macroscopically a dark, brownish discoloration has been present, while microscopically extensive hemosiderin has been described. In addition, thickened leptomeninges, neuronal loss of various degrees, reactive gliosis, and demyelination have been found Throughout our review, the presence of a recurrent hemorrhage is a must for SS development. However, there have been some etiological groups of pathologies causing the hemorrhage. Among the well-known causes, SS is most commonly caused by a spinal dural defect in 47% of cases. Tumors including ependymoma, oligodendroglioma, pineocytoma, and paraganglioma are responsible for 35% of SS. CNS tumor could be one of the sources of bleeding, both pre- and postoperatory. Vascular malformations including cavernous angioma, arteriovenous malformation, and aneurysm are responsible for 18% of cases of SS .The SS is often characterized by a triad of symptoms, sensorineural hearing loss (95%), cerebellar ataxia (88%), and pyramidal signs (76%) Superficial siderosis is considered a secondary effect instead of a real diagnosis. Therefore, the clinical symptoms and MRI findings may not correspond. MRI diagnosis can be made in the absence of symptoms Ideally, the treatment of the cause would interrupt the SS development. Some chelation therapies have been attempted. Deferiprone can be used safely with closed monitoring of possible side effects until the discovery of a new and effective option for the treatment of superficial siderosis Written informed consent for the publication of this case report was obtained from the patient.


Malformations of the bronchopulmonary foregut can lead to the formation of bronchogenic mediastinal cysts (BMC). BMC are rare congenital malformations usually found in the middle or posterior mediastinum. Only one-third of patients with BMC are symptomatic. We report a case of BMC in a 48-year-old female who was referred to the cardiothoracic surgeons due to an incidental finding of an anterior mediastinal mass on investigation for intermittent chest pain. The mass was treated surgically with a partial median sternotomy and mass excision. The patient’s symptomology and mass histology were atypical for BMC. At follow-up, the patient reported no residual symptoms. This case demonstrates the significance of considering BMC, especially the anterior subtype, as a differential diagnosis in the findings of patients with intermittent chest pain and computerized tomography findings of a mediastinal mass. Malformations of the bronchopulmonary foregut can occur in gestational weeks 4–6, which may lead to bronchogenic mediastinal cysts (BMC) , 2. BMC A 48-year-old female was referred to the cardiothoracic unit for a suspicious anterior mediastinal mass. Nine months prior, she presented to the emergency department with left-sided facial, upper and lower limb paraesthesia, pain, weakness and 2-week history of sore throat. She denied dysphasia or dysphagia. Neurological examination was normal and throat examination revealed tonsillar exudates. Non-contrast computerized tomography (CT) head noted no active disease. The patient was commenced on Penicillin V for tonsillitis and aspirin for suspected transient ischaemic attack (TIA). She was referred to the TIA clinic for further investigation where her National Institute of Health Stroke Scale score and Rankin score was 0, making vascular aetiology unlikely.Three months later the patient re-presented with intermittent chest pain associated with a 2-day history of non-productive cough. She was investigated with a chest X-ray and then a contrast CT chest  and 2. CA repeat contrast CT 5 months later confirmed no progression. However, the patient reported increasing chest pain and breathlessness and was referred for elective excision. The patient underwent partial median sternotomy to the fourth intercostal space, thymectomy and mediastinal mass excision. The aorta, brachiocephalic trunk, superior vena cava and thymus along with the superior and inferior cornu were dissected. The hard and nodular mass was situated on the right side of the anterior mediastinum and was adherent to the right upper lobe and phrenic nerve.Histopathology of the mass showed a unilocular cyst lined by ciliated and non-ciliated columnar to cuboidal epithelium with focal squamous metaplasia. The cyst wall showed focal hyaline fibrosis and calcification and contained smooth muscle, bronchial mucous glands and cartilage. There was patchy chronic inflammation with granular eosinophilic material and scattered cholesterol clefts on the cyst wall in keeping with BMC. The surrounding fatty tissue contained small lobules of benign thymic tissue showing minimal focal nodular epithelial hyperplasia, essentially a normal thymus. There was no evidence of granulomatous inflammation of malignancy.Post-operative recovery was uneventful, with no signs of infection. During the 2-week follow-up, the patient denied any shortness of breath or any restriction on activities of daily living.Bronchogenic cysts are rare congenital defects present due to the abnormal germination of the embryonic foregut that results in a fluid-filled pouch instead of normal bronchial development . Most coThe management of those with bronchogenic cyst relies on the symptoms of the patient. Thoracotomy or video-assisted thoracoscopic surgery can be used to resect cysts that are symptomatic. Management of asymptomatic cysts is debated, but surgical management is preferred . ThoracoNone declared.None.


Insulinoma is an uncommon insulin-secreting neuroendocrine tumor that presents with severe recurrent hypoglycemia. Although cases of extrapancreatic insulinomas have been reported, the majority of insulinomas occur in the pancreas. The number of reported cases of ectopic insulinomas with follow-up assessments is limited and they do not report disease recurrence. The current report presents the first documented case of recurrent extrapancreatic insulinoma with 8 years of follow-up, provides relevant literature review, and proposes surveillance and treatment strategies.We describe an insulinoma localized in the duodenal wall of a 36-year-old female who presented in 2013 with weight gain and Whipple’s triad and was successfully managed with duodenotomy and enucleation. She presented again in 2017 with recurrent Whipple’s triad and was found to have metastatic disease localized exclusively to peripancreatic lymph nodes. Primary pancreatic insulinoma was not evident and her hypoglycemia resolved following lymph node dissection. Eight years after initial presentation continuous glucose monitoring (CGM) showed a trend for euglycemia, and PET-CT Gallium 68 DOTATATE scan evaluation indicated absence of recurrent disease.Insulinomas are rare clinical entities and extrapancreatic insulinomas are particularly uncommon. Follow-up evaluation and treatment strategies for ectopic insulinoma recurrence presents a significant clinical challenge as the condition has hitherto remained undescribed in the literature. Available evidence in the literature indicates that lymph node metastases of intrapancreatic insulinomas likely do not change prognosis. Given the absence of long-term data informing the management and monitoring of patients with extrapancreatic insulinoma, we suggest patient education for hypoglycemic symptoms, monitoring for hypoglycemia with CGM, annual imaging, and a discussion with patients regarding treatment with octreotide or alternative somatostatin receptor analog therapies. Insulinoma is a rare neuroendocrine tumor (NET) with an incidence of 0.4 cases per 100,000 per year, and is typically seen in the 5th decade of life with a slight predominance of the female sex , duodenoIn the current report, we describe a 36-year-old female who presented with a primary insulinoma in the wall of the second portion of the duodenum that was surgically managed with resolution of her hypoglycemia. She then presented 4 years later with recurrent hypoglycemia and localized peripancreatic lymph node disease. We provide 8-year follow-up results and discuss strategies for follow-up evaluation, surveillance, and management of recurrent, metastatic extrapancreatic insulinoma.In June 2013, a 36-year-old female presented with Whipple’s triad  and weight gain of 40 pounds. Her serum glucose level was 49 mg/dL  with inappropriately elevated insulin level of 8.1 μIU/mL  and C-peptide level of 2.2 ng/mL  prior to receiving dextrose. A 72-hour fast confirmed hyperinsulinemic hypoglycemia at 26 hours . Serum calcium level was 8.8 mg/dL [normal range: 8.6–10.2 mg/dL] and parathyroid hormone level was 54 pg/mL [normal range: 10–65 pg/mL].Abdominal CT with intravenous contrast showed a 1.1 cm × 1.6 cm × 2 cm hypervascular mass in the second portion of the duodenum without obstruction and a normal pancreas. OctreoScan did not show uptake in the area of the mass. Esophagogastroduodenoscopy identified a mass in the duodenal sweep and endoscopic ultrasound (EUS) showed a 1.6 cm × 1.6 cm duodenal mass  showed no glucose values < 60 mg/dL, and CT of the abdomen with intravenous contrast did not show evidence of disease. In 2015, Ga-68-DOTATATE imaging was not yet available at our institution to allow for further imaging evaluation.In July 2017 the patient’s hypoglycemic symptoms returned with increasing frequency. Placement of another 7-day CGM showed that 29% of glucose values were < 60 mg/dL and revealed a pattern of nocturnal hypoglycemia. Abdominal CT evaluation did not show evidence of disease. PET-CT with Ga68-DOTATATE showed a 1.5 cm × 1.3 cm soft tissue nodule adjacent to the inferior pancreatic head and wall of the 2nd portion of the duodenum with maximum standardized uptake value (SUV) of 41 , single, sporadic, and arise within the pancreas with equal intra-organ distribution , 9, 10. Ectopic insulinomas usually develop in ectopic pancreatic tissue and have been reported in 0.5–15% of autopsies as well as in 1 out of 500 abdominal surgeries . In our A population-based study of 224 patients with surgically confirmed insulinomas presenting between 1927 and 1986 showed recurrence rates of 7% in patients with sporadic insulinomas compared with 21% in patients with MEN syndrome type 1 . RecurreIn patients with a biochemical diagnosis of insulinoma, surgical cure rates range from 77 to 100%. When technically possible, tumor enucleation is preferred. For tumors not amenable to enucleation, various surgical techniques can be pursued, such as segmental resection of the pancreas, distal pancreatectomy, or pancreaticoduodenectomy . ComplicMalignant insulinomas are those that show evidence of local invasion into surrounding soft tissue or distant metastases to the liver or lymph nodes. The 10-year survival for malignant insulinomas is reported to be 29% . AggressEverolimus, an oral mTOR inhibitor, has been used for malignant insulinomas associated with refractory hypoglycemia . The exaAs improvements in imaging studies continue to increase the frequency of pancreatic NET detection, recent studies have also evaluated the role of lymph node metastases in prognosis and management strategies. A retrospective review found tumor location, tumor size, Ki-67 index, and presence of lymphovascular invasion were associated with lymph node metastasis. Tumor diameter > 1.5 cm, tumors located in the head of pancreas, presence of lymphovascular invasion on surgical pathology, and Ki-67 index > 20% were reported to frequently present with lymph node metastasis . FurtherAt the present time, our patient continues to do well clinically with no evidence of significant hypoglycemia. She has declined octreotide treatment due to concerns with side effects and wishes to continue active surveillance with imaging. Given her recurrence of hypoglycemic symptoms and evidence of lymph node metastases 2 years after initial enucleation surgery, we suspect that she has microscopic, undetectable, clinically silent disease. As such, we will continue to monitor her closely for redevelopment of hypoglycemia.Extrapancreatic insulinomas are rare and the few cases of the tumors described in the literature do not report long-term follow-up assessment. This report presents the first known case of an extrapancreatic insulinoma recurrence and the longest reported followup. Current evidence shows that lymph node metastases of intrapancreatic insulinomas likely do not change prognosis. It is unclear if this is applicable to extrapancreatic insulinomas as well. More long-term outcome data are necessary to help determine how these patients should be monitored and managed. To monitor and manage extrapancreatic insulinoma recurrence we suggest patient education for hypoglycemic symptoms, monitoring for hypoglycemia with continuous glucose monitoring, annual imaging, and an ongoing discussion with patient regarding treatment with octreotide or alternative somatostatin receptor analog therapies.


Cystitis glandularis is a common bladder epithelial lesion characterized by hyperplasia and metaplasia of the bladder mucosa epithelium. The pathogenesis of cystitis glandularis of the intestinal type is unknown and less common. When cystitis glandularis  is extremely severely differentiated, it is called florid cystitis glandularis (the occurrence is extremely rare).Both patients were middle-aged men. In patient 1, the lesion was also seen in the posterior wall and was diagnosed more than 1 year ago as cystitis glandularis with urethral stricture. Patient 2 was examined for symptoms such as hematuria and was found to have an occupied bladder; both were treated surgically, and the postoperative pathology was diagnosed as florid cystitis glandularis , with mucus extravasation.The pathogenesis of cystitis glandularis  is unknown and less common. When cystitis glandularis of the intestinal type is extremely severely differentiated, we call it florid cystitis glandularis. It is more common in the bladder neck and trigone. The clinical manifestations are mainly symptoms of bladder irritation, or hematuria as the main complaint, which rarely leads to hydronephrosis. Imaging is nonspecific and the diagnosis depends on pathology. Surgical excision of the lesion is possible. Due to the malignant potential of cystitis glandularis of intestinal type, postoperative follow-up is required. Cystitis glandularis (CG) is a proliferative disease of the bladder mucosa characterized by transitional cell glands. CG was first described by Morgagni et al. in 1761 . He had a history of hypertension. There was no history of a long-term indwelling urinary catheter, no history of long-term toxic exposure, and the rest was not exceptional. Urine routine showed erythrocytes 7,468/μL. Urine microscopy showed erythrocytes ++++ (>100)/HP. CT scan showed thickening of the right bladder wall. Contrast-enhanced CT scans showed enhancement. After adequate preoperative preparation, resection of the bladder mass was performed. Intraoperatively, the lesion was seen to be located in the bladder triangle and at the opening of the right ureter. The basal width of the lesion was approximately 3 cm × 3 cm, and no blood spray was seen at the left ureteral opening. Due to the wide resection of the right ureteral opening, one F6 ureteral stent tube was placed in the right ureter. The postoperative naked eye view is grayish-gray-brown tissue. The differentiation from von Brunn's nest was it was more dense and irregular, with different morphology, distinct atypical hyperplasia, significantly enlarged lumen, and mucus lakes in the interstitium. The pathological diagnosis was florid cystitis glandularis  with mucus extravasation. Immunohistochemistry showed the following: GATA3 (+), CK7 (+), CDX-2 (+), and Ki-67 (10%)  the embryonic origin theory: during bladder development, ectopic embryonic remnants due to congenital anatomical or abnormal factors can form glands, leading to cystitis glandularis; (2) the theory of epithelial transformation: under long-term pathogenic factors, it can lead to the transformation of the uroepithelium into the glandular epithelium and intestinal epithelial metaplasia; (3) epithelial cells undergo degeneration to an already differentiated previous stage . The mosWhen cystitis glandularis  is extremely severely differentiated, it is called florid cystitis glandularis (the occurrence is extremely rare). It is more common in women than men, and cystitis glandularis of the intestinal type is more common in the bladder neck, and trigone. The clinical manifestations are mainly symptoms of bladder irritation or hematuria as the main complaint, which rarely leads to hydronephrosis , 6. All The formation of von Brunn's nests was visible on microscopic examination in all cases in this group, along with extravasation of mucus and extremely severe differentiation. This is consistent with previous literature reports. Ultrasound, CT, and MRI can all detect lesion bladder wall thickening. It can be found with a nodular or diffuse appearance . ImagingHowever, cystitis glandularis is not a high risk factor for adenocarcinoma and uroepithelial carcinoma. The natural history of this disease is unknown. Both patients were treated surgically and had a good postoperative prognosis.The pathogenesis of cystitis glandularis  is unknown and less common. When cystitis glandularis  is extremely severely differentiated, it is called florid cystitis glandularis (the occurrence is extremely rare). It is more common in women than in men, and cystitis glandularis of the intestinal type is more common in the bladder neck and trigone. The main clinical manifestations often include hematuria and bladder irritation. However, it rarely leads to hydronephrosis. The presentation is often nonspecific and pathology is the best diagnostic modality. Surgical excision of the lesion is possible. The differentiation from von Brunn's nest is it is more dense and irregular, with different morphology, distinct atypical hyperplasia, significantly enlarged lumen, and mucus lakes in the interstitium. Due to the malignant potential of cystitis glandularis of the intestinal type, postoperative follow-up is required.


Craniofacial involvement in fibrous dysplasia is a rare occurrence and complications of it are even rarer. Involvement of the sinonasal region can pose a challenge in both diagnosis and management. We hereby present a case of craniofacial fibrous dysplasia complicated with secondary aneurysmal bone cyst formation in a 15-year-old male patient. Fibrous dysplasia (FD) is a benign tumor-like disorder of the skeletal system which can affect virtually any bone in the body, representing approximately seven percent of this subgroup . CharactComplications are not uncommon in FD since the affected bones are rendered weak and prone to pathological fractures. Aneurysmal bone cysts (ABC) are rare benign tumor-like lesions of vascular origin affecting typically involve the long bones of the extremities , membranA 15-year-old male patient, previously diagnosed with craniofacial FD, presented with a rapidly developing mass lesion involving the nasal cavity with proptosis of the left eye. There was no history of epistaxis. Clinical examination revealed facial dysmorphism and proptosis of the left eye. Endoscopic examination of the nasal cavity shows a friable reddish mass lesion involving the bilateral nasal cavity. Clinical differentials included vascular mass lesions, i.e, hemangioma or juvenile nasopharyngeal angiofibroma. The patient was advised of Magnetic Resonance Imaging (MRI) for further characterization.On MRI, a large multi-lobulated lytic expansile lesion with peripheral T1/T2 hypointense component was seen involving the nasal cavity causing its expansion  and lymphoma. A complete imaging workup is necessary not only to delineate and characterize the primary pathology but also the complications. Radiographs of the skull and paranasal sinuses can be initially done, however, does not provide much information. CT is useful for the assessment of the structure of affected bone and complications like pathological fractures, while MRI is an excellent modality for the assessment of intraorbital or intracranial extensions of the lesion. Most benign etiologies such as polyps are solid lesions with associated features of sinusitis,i.e., mucosal thickening and thickening of sinus walls. While the malignant masses are most destructive, invading various surrounding structures. Vascular tumors like JNA will show avid enhancement with multiple arterial feeders identified on angiography. Cystic nasal masses in children include congenital entities such as congenital nasolacrimal duct mucocele, dermoid cyst, and encephalocele, inflammatory and infectious lesions such as mucoceles and polyps, benign or malignant neoplasms with internal areas of degeneration and necrosis. Multiloculated cystic lesion with fluid-fluid levels is a characteristic imaging feature of ABC which was the clincher in our case. Further, the assessment of surrounding structures such as nerves or bones can provide a clue to the origin of the lesion. In our case, the surrounding bone showed expansion with the ground glass matrix suggestive of FD.FD can often be complicated by pathological fractures. Secondary development of ABC in FD is a rare occurrence, with only 15 cases reported so far -8. Of thThe concomitant occurrence of FD and ABC was first described by Branch et al.  in 1986.En-bloc resection with bone grafting is the treatment of choice in most cases . ComplexThis case represents a common condition with an uncommon complication and clinical presentation. Sino-nasal mass in a young patient can have several differential diagnoses, ranging from congenital, inflammatory, and infective etiologies to neoplastic processes. A pre-treatment cross-sectional imaging can adequately characterize and delineate these lesions as well as guide the treatment options. Adequate anatomical knowledge with a mind open to the possibility of lesions arising from different tissues, i.e, sino-nasal mucosa, bony structures, respiratory epithelium, vessels, and lymphatics, present in this region is necessary to evaluate masses of this complex anatomical site. Also, assessment of lesions as well as surrounding structures is necessary for guiding the pre-operative and operative management of the patient.