Meigs syndrome is a rare disorder characterized by a triad of benign ovarian tumor, ascites, and pleural effusion. Despite its benign nature, its presentation can be similar to metastatic malignancy, making preoperative diagnosis often challenging. The differential diagnosis includes serious and even life-threatening conditions. Meigs syndrome is most common in postmenopausal women and is extremely rare in children. It is often undiagnosed until an invasive surgery is performed. The fact that surgery includes a unilateral salpingo-oophorectomy in females of reproductive age raises concerns for future fertility and other risks associated with this intervention. Familiarity of radiologists and pediatric surgeons with clinical and imaging findings is beneficial to improve preoperative planning, thereby minimizing invasive surgery and preserving ovarian tissue. Meigs syndrome is a rare disorder  comprisiDespite its benign nature, it is often undiagnosed until an invasive surgery is performed. The surgery typically includes a unilateral salpingo-oophorectomy in females of reproductive age . This raA previously healthy premenarchal 12-year-old female presented with acute abdominal pain associated with a three-month history of abdominal distention and unintentional weight loss of 3 kg. Physical examination revealed a shifting dullness suggestive of ascites and a palpable mass in the pelvis.Laboratory workup showed cancer antigen 125 (CA-125) to be elevated to 1,160 U/mL with normal carcinoembryonic antigen, cancer antigen 19-9, alpha-fetoprotein, beta-human chorionic gonadotropin, and lactate dehydrogenase. Ultrasound revealed a large left adnexal mass measuring 14 × 12 × 6 cm with significant ascites. Computed tomography of the chest, abdomen, and pelvis showed mild-to-moderate right pleural effusion and a large heterogeneously enhancing complex cystic mass arising from the left ovary measuring 14.8 × 12.5 × 6 cm, along with large ascites , and conserving both ovaries should be a priority in an expected benign course . UnilateTo confirm Meigs syndrome, postoperative resolution of ascites and pleural effusion and histological confirmation of the tumor are usually required .We present a case of Meigs syndrome in a pediatric patient, a rare condition in a rare age group for such a diagnosis. This benign disease must remain on the differential diagnoses for every female presenting with the classic triad because recognizing Meigs syndrome early in the workup course allows for less aggressive intervention compared to cases of malignancy. The main aim must be preserving ovarian tissue, particularly in the reproductive age group.


The afferent visual system is one of the most common structures involved in patients with craniopharyngioma, and the manifestations include deficits in visual acuity, color vision, and visual fields. Here, we report a case of craniopharyngioma that presented with acute blindness in an elderly man. A healthy 54-year-old man presented with an acute progressive blurring of vision and became blind in six weeks. He developed symptoms of increased intracranial pressure only a week after becoming blind. On examination, visual acuity in both eyes was no perception of light (NPL). He also had left esotropia with restriction of left eye abduction. Both pupils were not responsive to light. The anterior segment was normal in both eyes. Fundoscopy showed bilateral pale optic discs. Computed tomography scan and magnetic resonance imaging revealed a suprasellar mass consistent with craniopharyngioma that compressed the optic chiasma and adjacent brain structures with the presence of hydrocephalus. He underwent uneventful tumor debulking surgery. However, his vision remained NPL postoperatively. Ocular manifestations could be the only symptoms in craniopharyngioma. The delayed presentation may lead to a guarded prognosis. Craniopharyngiomas are generally benign in nature . HoweverPart of this article was previously presented as a poster at the 35th Singapore-Malaysia Joint Meeting in Ophthalmology from January 17 to 19, 2020, in Singapore.A healthy 54-year-old man presented with an acute progressive blurring of vision in both eyes. It started with generalized blurring and he became totally blind within six weeks. He experienced horizontal diplopia before total blindness. Otherwise, there was no eye pain, eye redness, or proptosis. He started to develop headaches only one week after becoming blind. The headache was described as frontal, throbbing in nature, and aggravated on straining. However, there was no nausea or vomiting. He also had lethargy, loss of appetite, and loss of weight. His ocular history was unremarkable. On examination, the visual acuity in both eyes was no perception of light (NPL) in all four quadrants. The anterior segment in both eyes was normal. Bilateral pupils were dilated 5 mm in diameter and not responsive to light. The intraocular pressure (ICP) was normal in both eyes. There was left esotropia on the primary gaze with restriction in abduction. Fundoscopy examination revealed a pale optic disc in both eyes. Computed tomography (CT) scan and magnetic resonance imaging (MRI) revealed a suprasellar mass consistent with craniopharyngioma that compressed the optic chiasma and adjacent brain structures with the presence of hydrocephalus Figures , 2. He uCraniopharyngiomas are rare tumors, with the overall incidence ranging from 0.12 to 0.13 per 100,000 per year . They coAs for the clinical features, they are dependent on the size, location, and growth of the tumor, as well as the relationship with the surrounding structures. The most common presentation is visual disturbance, and studies have shown that the prevalence of visual disturbance could be as high as 65-82% ,11,12. SOn the other hand, symptoms of increased ICP are very common and have been reported in 75% of cases. However, cranial nerve palsy is not as common with an 11% prevalence in the same study . Our patRegarding diagnosis, any suspicion should be based on clinical and radiological findings. CT and/or MRI are the choices for radiological investigation. CT has better visualization of calcifications and bony structures, whereas the assessment of tumor morphology and relationship with surrounding structures is better achieved by MRI. Therefore, MRI is the imaging of choice in preparation for surgery . HydroceSurgery remains the first-line management. Complete resection is the goal of surgical treatment. Surgical complications might occur if the tumor size is too big or if the tumor is in close relation to other vital structures. Partial resection would be favorable if complete resection is not possible. Radiotherapy is the treatment of choice if the patient is not suitable for surgery, in cases with incomplete tumor resection, or in recurrent cases. Chemotherapy is another treatment option, especially for cystic tumors . Visual Ocular manifestations of rapid progression can develop much earlier than other symptoms in craniopharyngioma. Differential diagnosis of any suspicious ocular feature should not be limited to just ocular in origin as it could be the only or earlier symptom or sign. A high index of suspicion and timely imaging can be vision or lifesaving.


Angiofibroma is an uncommon, highly vascular benign lesion that occurs in the head and neck region, typically arising in the nasopharyngeal area, potentially with a locally aggressive course. Angiofibroma with a primary intraoral presentation is extremely rare; few case reports have been published in the literature, with only three cases of angiofibroma in the mandible published to date. In this case, a 37-year-old man presented with swelling at the right mandible and underwent enucleation of the lesion under general anesthesia. After 1-year follow-up, there were no signs of recurrence. Angiofibroma is a rare, benign, highly vascularized tumor that commonly presents during male adolescence. Its origin is unknown . It typiThe patient  presented with swelling of the right posterior mandibular area. He had a clear medical history. He complained of a small, dark blue-colored localized swelling at the area of the attached gingiva between teeth numbers 45 and 46 for 24 days before he went to a general dentist. An antibiotic was prescribed for five days, but his condition did not improve, so the dentist decided to extract tooth number 46. After extraction, the swelling started to gradually increase until it occupied the buccal vestibule. He noted pus and bleeding from the mass. Extraoral examination revealed significant swelling on the right side of the face that extended from the inferior border of the mandible to the level of the zygoma . SwellinThe patient had no history of paresthesia related to the inferior alveolar or lingual nerve. Intraorally, a huge mass was found to occupy the right buccal vestibular area. It was firm, measured 5 × 5 × 3 cm, red in color, covered with a whitish film, and pedunculated from the extraction socket, with ulceration noted at the level of the occlusal table due to mastication . OrthopaIncisional biopsy was performed, and the extraosseous portion was excised and sent for histopathological assessment. Bleeding was encountered during the biopsy, which was controlled by local measures (Surgicel and suture). At the 1-week follow-up visit, the mass had grown again and reached its previous size.Histopathological findings were suggestive of angiofibroma. Microscopy revealed an ulcerated, markedly inflamed squamous epithelial mucosa with underlying proliferation of fibrous tissue with interspersed abundant vascular channels. The vascular channels had architectural variability from the small capillaries to the sinusoidal ectatic vessels, to the larger vessels of venous type with distinct muscle layer. The fibrous stroma consisted of abundant collagen fibers of variable density and stellate or ovoid spindle cells with a fibroblastic/myofibroblastic appearance .Computed tomography (CT) angiography was performed to assess lesion vascularity. No obvious large vascular channel supplying the lesion was detected. A large venous tributary in the vicinity of the lesion was observed to be draining into the right external jugular vein. A bony perforation at the lingual side was associated with enhanced soft tissue component.The decision was made to perform enucleation of the lesion under general anesthesia. The patient was in the supine position during the operation and received oral intubation; hypotensive general anesthesia was applied. Excision of the extraosseous part was performed along with enucleation of the intraosseous lesion. The surgery was uneventful, and the lesion was easily detached from the bone with no adhesion noted. Encountered bleeding was controlled using cautery and bone wax. The wound was closed primarily. The final histopathological report confirmed the presence of angiofibroma and provided the same histopathological description. Immunohistochemical analysis using different stains has been performed to confirm the diagnosis. The lesional cells were positive for vimentin, smooth muscle actin, and Bcl2 and negative for Ck AE1/AE3, desmin, CD34, and S100Prot Figures .The patient received regular follow-up . Healing was within normal limits, and no recurrence of overgrowth, swelling, or discharge has been observed .At 1-year follow-up, CT with contrast and CT angiogram were performed to confirm no recurrence and no arterial or venous regrowth. The report concluded the presence of bone defect with no soft tissue component and no abnormal arterial or venous enhancement. Comparing the preoperative CT with the 1-year postoperative CT, bone regeneration was noted, and only a small portion of the inferior border defect remained .The significance of this report lies in the rarity of the presence of angiofibroma, which commonly occurs in the nasopharynx, in the mandible.The etiology of angiofibroma is unclear. Theories about the origin and histogenesis of angiofibroma vary  .Angiofibroma are rare tumors of the maxillofacial region, accounting for 0.05% of head and neck tumors . The preIt is suspected that ectopic tissue may be located farther away from the usual place and may have been the cause of the extranasopharyngeal location . HistoloNasopharyngeal angiofibromas in the head and neck area usually arise in adolescent males . In theiDepending on the size and location of the angiofibroma, different treatment modalities are available. Most physicians agree that surgery is the primary treatment modality for early-stage disease . PreoperIn our case, no embolization was planned, as there was no obvious large vascular channel supplying the lesion. No serious bleeding was encountered during the operation. Some authors have reported the successful surgical treatment of juvenile angiofibroma with acceptable blood loss without embolization , 5, 14.Some researchers advised resection to prevent recurrence , 8. MostOther treatment modalities include radiation therapy, cryotherapy, hormone therapy, embolization, arterial ligation, and sclerosing agent use . May et The significance of this report lies in the rarity of the presence of angiofibroma, which commonly occurs in the nasopharynx at young age, in the mandible. Although exceedingly rare, angiofibroma should be considered one of the differential diagnoses of a lesion that arises in the mandible.


The metanephric adenoma is an extremely rare, benign, embryonal-epithelial neoplasm of the kidney and has a good prognosis with appropriate treatment. It can present at any age and is often asymptomatic. Histologically, the lesion is well established; however, there have been only a few cases described with available detailed imaging findings, most of them with large renal masses typically depicted by computed tomography (CT). This case report includes imaging of contrast-enhanced MRI, contrast-enhanced ultrasound (CEUS), and CT, and thus adds to the information available, potentially promoting a nephron-sparing clinical pathway. We report on the clinical presentation, imaging, histopathological diagnosis, and treatment data of a 27-year-old female, in whom an incidental, symptomatic kidney tumor was detected. CT, CEUS, and MRI showed a suspicious unifocal renal lesion with inhomogeneous enhancement, which was indistinguishable from renal cell carcinoma. After laparoscopic resection, a metanephric adenoma with microscopically partially glandular, partially nest-like solid growth and without distinctive atypia was diagnosed pathohistologically. Immunohistochemistry results were positive for Wilms Tumor 1 and CD57 and negative for EMA and CK7: 2–3% positive cells in MIB1 coloring. At 3-month and 1-year follow-up, the patient was asymptomatic and imaging showed no recurrence of renal masses or metastases. The metanephric adenoma (MA) is a rare embryonal epithelial tumor of the kidney. It was first described in 1979 by Bove  and showWe report a case of histologically confirmed MA in a 27-year-old female and describe its appearance on MRI (magnetic resonance imaging), CEUS (contrast-enhanced ultrasound) and CT (computed tomography). As seen in this case and according to the experiences of others in the literature, it is very challenging to make a definite diagnosis based on imaging, and therefore, pathohistological correlation by nephron-sparing surgery is needed.A 27-year-old woman presented to our regional tertiary hospital due to spiking fevers of unknown origin and occurring every 8 weeks over the past 12 years. At primary presentation in 2012, there were no features of vasculitis, collagenases, or mutation of the MEFV-Gene . Initial abdominal sonography and laboratory tests were normal. Apart from a known hypothyroidism and a previous inguinal hernia repair, the patient was healthy. Her family history was unremarkable.The patient presented again in late 2019 in the rheumatology department of our university hospital. The periods of spiking fevers had shortened in duration in the past 5 years, from 7 to 3 days, and become less severe. Often, they occurred with a sore throat, body aches, or abdominal pain. The patient was also a healthcare professional and noted a concordance with high stress levels.2). Rheumatologic laboratory was non-conclusive. The patient did not have an elevated temperature on the day of presentation and showed to be in a good general condition without hematuria, frequent urination, odynuria, and dysuria.After a thorough but unremarkable clinical examination, blood laboratory was ordered, which showed a slightly low hemoglobin level of 11.2 g/d, lowered erythrocytes with 3.6 Mio/μL, and slightly elevated lymphocytes with 47.3% with at the same time slightly decreased absolute neutrophil counts of 1.8 thousand/μL. Otherwise the results were normal: electrolytes as well as renal specific laboratory parameters were normal . In early December 2019, a solid mass was therewith found in the cortex and parenchyma of the lateral midportion of the left kidney. The lesion was well circumscribed, homogeneous hypodense in comparison to the contrast-enhanced parenchyma and measured approximately 12 mm × 11 mm × 11 mm. The density was approximately 90 Hounsfield Units (HU). For imaging details, please see The patient presented again a week later at our department for the CEUS . The lesion in the left kidney was still detectable with well-circumscribed borders, a maximum diameter of 12 mm, and was slightly inhomogeneous with a low echo intensity in comparison to the surrounding tissue, see In the following week, the patient was assessed in supine position on a 1.5T MRI scanner  with a 64-channel body array coil and a protocol consisting of T1w with additional in- and opposed phase images, T2w, diffusion-weighted and Gadobutrol enhanced T1w imaging in arterial, portal venous, early and late venous phases.In keeping with the previous imaging, the renal mass was well-demarcated without a fibrous capsule with a slight bulge of the intact renal outline. In the T2-weighted sequence the lesion showed a hypointense signal in relation to the surrounding renal cortex and medulla A. In the®, Bayer, Leverkusen, Germany) contrast-enhanced fat saturated T1-weighted images showed a slight heterogeneous enhancement of the lesion, especially in portal venous and venous phases, with an indicated wash-out kinetic in late venous phase , could not be excluded.Consequently, the patient received a robot-assisted (Davinci) laparoscopic partial nephrectomy of the left kidney in March 2020. Intraoperative sonography was conducted, and the intraoperative specimen revealed a slight buckling of the tumor at the renal cortex and a faintly brown coloring. The lesion did not show a communication to the collective system and was removed without complication. At the first follow-up 3 months after surgery, ultrasound showed normal postoperative findings. At 12-month follow up, the patient was clinically symptom-free (without the undulant fever) and in non-enhanced MRI there was no renal recurrence detectable .The macroscopic findings of the pathohistological report were a completely imbedded, brownish tumor, which measured 17 mm × 6 mm × 6 mm. Microscopically, the tumor cells were partially glandular, partially nest-like with solid growth and without distinctive atypia. The tumor cells were middle-to-large with a narrow cytoplasm and a large cell nucleus. There was chromatin homogeny and overall low pleomorphia. Immunohistochemistry results were positive for Wilms Tumor 1 and cytoplasmic CD57 and negative for epithelial membrane antigen (EMA) and CK7; 2–3% positive cells in MIB1 coloring. The pathological diagnosis was MA of the left kidney without an indication for malignancy.The metanephric adenoma is a rare epithelial tumor derived from metanephrogenic embryonic tissue, which can occur at any age but has a diagnosis peak in the fifth or sixth decade of life and a female predominance of 2:1 ,9,10. ItOn imaging, MA presents as a well-defined, round or oval, soft tissue lesion with often bulging of the outer cortex contour. Most MA show to be non-encapsulated, conversely most PRCC are bordered by a thick fibrous pseudo-capsule . On ultrCT typically reveals a well-defined, non-invasive, solid, and mostly unilateral and unifocal lesion, which can include calcifications (20%), cystic spaces and necrosis ,18. MA vIn this case, MA appeared as a well-demarcated soft tissue mass with contrast sparing compared to the enhanced adjacent renal parenchyma/cortex. There were no calcifications or cystic transformations apparent. Contrast media uptake could not be interpreted due to the lack of unenhanced imaging or dual energy application to reconstruct virtual monoenergetic reconstructions.There are no distinct attenuation patterns described for MA in CT. According to the literature, MA can be depicted as a slightly hyper- or hypodense lesion compared to the underlying parenchyma and can show enhancement in dynamic CT examinations. However, the enhancement degree is lower than the renal cortex in both the corticomedullary and nephrogenic phase with, at times, a gradual and prolonged enhancement ranging from primarily peripheral to centripetal enhancement ,20,21.Such non-specific characteristics are also seen in MRI; however, this imaging technique often cannot further elucidate the diagnosis. MA has been previously described as a hypointense mass in T1 weighted imaging with iso- or slightly hyperintense imaging characteristics on T2 weighted imaging ,22. In kImaging is inadequate to differentiate MA from other renal masses because specific imaging characteristics do not exist. Pathological diagnosis is definitive but pre-operative biopsy uncommon.Macroscopically MA is described as a solitary, well defined tumor with varying size, coloring (from grey to tan to yellow) and tissue stiffness (soft or firm) . As discMicroscopically, MA is composed of tightly packed small and uniform  cells, with scanty cytoplasm, arranged in solid sheets or small acini-like aggregates, minimal intercellular stroma with various degrees of intra-tumoral fibrous bands, absence of mitoses and intermingled with glomerular or papillary architectural patterns ,3. PsammAdditionally, immuno-histochemical characterization usually shows an expression of CD57, often CK7 cytoplasmic immunoreactivity  and WT1 without showing epithelial membrane antigen (EMA) or AMACR ,19,22,23Genetic evidence suggests that metanephric adenoma is its own entity and can be differentiated from Wilms’s tumor, which shows an allelic imbalance at chromosome 11p13 . There iThis case has presented comprehensive radiological characteristics of MA including CEUS, CT, and MRI features with correlation to histopathology. It was depicted as a well-circumscribed, heterogeneous renal mass protruding the renal outline with a slight diffusion restriction and a subtle contrast media uptake, which however, was less than the underlying parenchyma/cortex.MA should be considered in the differential diagnosis of malignant renal tumors, even though it is a rare entity. Since MA does not have specific clinical symptoms nor clear imaging characteristics that allow for unambiguous diagnosis, a differentiation of MA and other renal masses remains challenging. Due to the lack of differentiation from RCC, operation or biopsy should be performed. Likewise, patient management should not differ from that of RCC until histological confirmation of MA. However, isolated metastatic spread of MA has been reported and a consensus in follow-up imaging recommendation would be desirable.


Retroperitoneal extension is a rare and fatal complication of Fournier's gangrene (FG) which mandates immediate surgical intervention for better outcome.A 70-year-old male presented to the emergency department with a history of bilateral painful scrotal swelling for 7 days with fever and abdominal pain for 3 days. On his general examination, he was septic with necrotic patches in the perineum and bilateral scrotum. Imaging revealed soft tissue gas and collections in scrotum extending to the right retroperitoneum with massive collection suggestive of retroperitoneal abscess. Following resuscitation and intravenous antibiotics, immediate exploratory laparotomy was done to drain the retroperitoneal abscess followed by debridement of Fournier's gangrene. The patient remained well on follow up.Fournier gangrene is a fulminant polymicrobial infection of the perineum, scrotum and penis which when complicated by retroperitoneal extension, has a very high mortality. Majority of patients have an immunocompromised condition. Early diagnosis with prompt surgical drainage and debridement (within 6 hours) significantly reduces the mortality.High index of suspicion, careful clinical examination and timely use of imaging is crucial for early diagnosis of this rare but fatal complication of FG. Furthermore, adequate resuscitation with prompt surgical intervention is the key for a favorable outcome. •Fournier's gangrene (FG) is a type of necrotizing fasciitis involving the perineum and genital region.•Retroperitoneal extension of FG is a rare and fatal complication which is usually seen in immunocompromised patients.•Retroperitoneal abscess should be suspected in patients with FG who present with abdominal pain. There istridium . The infstridium . Howeverstridium . The clistridium . This sustridium .There have been very few cases reported of FG with retroperitoneal extension. We present here a rare and potentially fatal case of FG with retroperitoneal extension who was successfully managed with timely surgical debridement of FG and drainage of retroperitoneal abscess. This case report is presented in accordance with the SCARE criteria .2We reported this case following the updated consensus-based Surgical Case Report (SCARE) guidelines .3A 70-year-old retired army serviceman with no known comorbidities, referred from Nepalgunj Military hospital, presented to our emergency department with complaints of pain over the right lower abdomen for the last 3 days and painful bilateral scrotal swelling for 7 days. The abdominal pain was dull aching in nature, initially localized to the right lower abdomen but had progressively radiated to pelvic and bilateral flanks with no known aggravating factors on the day of presentation. There was no history of nausea, vomiting, loose stool or burning urination. His bowel and bladder habits were normal. He was managed in Nepalgunj Military hospital with oral antibiotics and analgesics for painful scrotal swelling. However, his symptoms further worsened followed by pain in abdomen and fever for the past 3 days. His past medical and surgical history, family history, drug and allergic history were insignificant. He is a non smoker and does not consume alcohol.He was advised for Ultrasonography (USG) abdomen and pelvis which showed right paracolic and paracaecal collection measuring 3 X 3 × 7 cm. With increased intensity of pain in the abdomen, he was further advised for Contrast Enhanced Computed Tomography (CECT) abdomen pelvis to confirm the diagnosis. CECT scan of abdomen and pelvis revealed soft tissue gas in the scrotum and retroperitoneal cavity. It revealed an ill-defined collection measuring 10 X 5 × 4 cm (1040 ml) in size is noted in right abdominopelvic cavity, extending superiorly up to subcostal region with extension to right lumbar, right paracolic gutter and right iliac fossa. Right kidney was displaced anteriorly by the collection suggesting retroperitoneal extension of the collection. Multiple interspersed air foci were noted in the collection extending to the scrotal wall mainly to the right side suggesting the retroperitoneal extension 3 cells/μL with 62% neutrophils and platelet count of 124 × 103 cells/μL (thrombocytopenia). The biochemical tests showed a creatinine: 1.3 mg%, total bilirubin: 1.5 mg%, direct bilirubin: 0.2 mg%, protein: 5.4 g%, albumin: 2.6 g%, Aspartate Aminotransferase (AST): 128 IU/L, Alanine Aminotransferase (ALT): 127 IU/L, Alkaline phosphatase: 127 IU/L. His Random Blood Sugar (RBS) was 128 mg/dl. The serology was non reactive for HIV, Hepatitis B and Hepatitis C. On radiological examination, X-ray abdomen erect/supine were unremarkable. A provisional diagnosis of retroperitoneal abscess secondary to Fournier's gangrene with sepsis was made and broad spectrum antibiotics were started.On his arrival to the emergency department, he was febrile (101 °F) and in acute distress due to pain with tachycardia and tachypnea. His vitals were suggestive of septic shock with blood pressure of 80/40 mmHg on presentation. However, after initial fluid resuscitation, his vitals were stabilized. The abdomen was distended and diffusely tender with localized guarding and rigidity on the right lower abdomen. On rectal examination, normal tone, finger stained with stool and no palpable mass were noted. On local examination of the scrotal wall, there was edema with erythema, and patches of blackish discoloration involving both sides of scrotum. The erythema was extending to the right lower abdominal wall. His laboratory investigation revealed an elevated white blood cell count of 21.3 × 10The patient underwent exploratory laparotomy and debridement of FG on the same day. Intraoperatively, infected necrotic tissues and foul-smelling exudates were found in the right retroperitoneum space requiring a wide excision of the left psoas muscle, lateral abdominal wall, and Gerota's fascia of the right kidney. Approximately 700 ml of purulent collection was drained from right retroperitoneal space and pus was sent for culture and sensitivity. No intraperitoneal collection was noted intraoperatively. A temporary abdominal drain was placed in the right retroperitoneal space and the abdomen was closed after adequate Normal Saline (NS) lavage. This was followed by debridement of Fournier's Gangrene of bilateral scrotum 4Fournier gangrene (FG) is a fulminant polymicrobial infection of the perineum, scrotum and penis. There is progressive obliteration of subcutaneous arteries leading to gangrene of the subcutaneous tissue . The comRetroperitoneal abscess has an insidious onset with atypical presentation. The most common source of retroperitoneal abscess is accounted to be genitourinary, gastrointestinal infections, injuries, or malignancy of adjacent retroperitoneal or intraperitoneal organs. Retroperitoneal involvement is seen in less than 30% cases with FG . AccordiThe initial investigation to evaluate FG requires blood workup and imaging. There is an increase in white blood cells with a potential left shift, coagulopathy, hyponatremia and elevated inflammatory markers (C-reactive protein and/or erythrocyte sedimentation rate). The elevation in serum Creatine Kinase and AST levels suggest a deeper infection involving the fascia which also helps differentiate Necrotizing Soft Tissue Infections (NSTI) from cellulitis . The wouIn addition to laboratory markers and imaging, various tools have been recognized in aiding the diagnosis of NSTI. The most frequently used tools with high diagnostic value are Laboratory Risk Indicator for Necrotizing Fasciitis (LRINEC) score, NSTI Assessment Score (NAC) and Uludag Fournier's Gangrene Severity Index (UFGSI). The UFGSI is considered to be a powerful indicator in regards to the 1995 Fournier's Gangrene Severity Index scoring system ,15.In suspicion of NSTI, a prompt surgical exploration with debridement and drainage along with antibiotic therapy is indicated in high risk patients (such as immunocompromised patients) . This ur5Retroperitoneal involvement with Fournier's Gangrene though rare should be suspected among patients complaining of abdominal pain. Timely diagnosis can be achieved by having a high index of suspicion, proper clinical examination and use of imaging, allowing prompt surgical intervention. For a favorable outcome, adequate resuscitation and IV antibiotics with prompt surgical drainage of retroperitoneal abscess and debridement of FG is crucial.The primary takeaway lesson from this case report is that the treating physician/surgeon should have a high index of suspicion for retroperitoneal abscess in FG who present with abdominal pain.Not required in our case.None.Sunil Basukala (SB) = Conceptualization, Supervision, Sunil Basukala (SB), Yugant Khand (YK), Soumya Pahari (SP) = Writing - original draft, Sunil Basukala (SB), Yugant Khand (YK), Soumya Pahari (SP), Kunda Bikram Shah (KBS), Aashish Shah (AS) = Writing - review & editing, All the authors read and approved the final manuscript.1.Name of the registry: Not applicable2.Unique Identifying number or registration ID: Not applicable3.Hyperlink to your specific registration (must be publicly accessible and will be checked): Not applicablesunil.basukala@naihs.edu.np.Sunil Basukala (SB), MD in Hospital Administration (MDHA), Master of Surgery (MS), Assistant Professor, Shree Birendra Hospital, Nepalese Army Institute of Health Sciences (NAIHS), 44600, Nepal. Email: 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.Not commissioned, externally peer-reviewed.All authors declare that they have no conflict of interest.


A 24‐year‐old right‐handed male presented with a 4‐month history of migraine of weariness. Typical accompanying symptoms with migraine like nausea, vomiting, photophobia and aura were not present. Non‐contrast CT brain revealed a left sided large frontotemporoparietal  arachnoid cyst. The patient underwent a cystoperitoneal shunt. Arachnoid cysts are benign lesions which account for 1% of all non‐traumatic intracranial masses. Their mechanism of formation is not completely understood and the natural case history is not well documented. Several patients may have no symptoms throughout their lives, while others may become symptomatic many years after the cyst is discovered.A 24‐year‐old right‐handed man with a 4 month history of migraine of weariness was referred to our outpatient by a neurologist. Migraine of weariness is characterized by fatigue and exhaustion to the point where the migraineur may feel nearly unable to respond to anything. He used to have extreme fatigue and exhaustion alongside unilateral headache. Nausea, vomiting, photophobia, and aura, which are common migraine symptoms, were not present. Neurological examination was unremarkable. Baseline investigations were normal. A left sided large frontotemporoparietal  arachnoid cyst was discovered on a non‐contrast CT scan of the brain Figure . A cystoAhtesham Khizar: Conceptualization; methodology; writing – original draft; writing – review and editing. Waleed Shahzad: Formal analysis; investigation; resources; software; visualization. Pradhumna Kumar Yadav: Formal analysis; methodology; supervision; visualization.The authors declare no conflict of interest.Not applicable.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Vitamin A is necessary for the function of retina, by the formation of pigments in rod and cone photoreceptors in the phototransduction cycle. One of the early ocular signs of vitamin A deficiency is nyctalopia. In prolonged untreated cases, other ocular signs including conjunctival Bitot's spots, corneal ulcer, and keratomalacia develop.We present a 30-year-old male who was admitted for evaluation by the rheumatologist due to oligoarthralgia. The patient had bariatric surgery for morbid obesity seven years earlier. The rheumatologist consulted us for the patient's foreign body sensation and night blindness complaints during the last one year.His ophthalmic examination revealed best-corrected visual acuity of 20/20 in both eyes by Snellen E-chart. Intraocular pressure was 12 mmHg and Ishihara test scores were 14/14 in both eyes. In slit-lamp examination, there was a reduction in tear meniscus height (5 mm) with bilateral Bitot's spots at the limbal conjunctiva [Figure 1].There were small yellowish punctate lesions in the peripheral retina [Figure 2A–B]. The optic nerve, macula, and vessels seemed normal [Figure 2C–D]. Peripheral spectral-domain optical coherence tomography (SD-OCT) showed sparse subretinal drusenoid deposit (SDD)-like lesions bilaterally [Figure 2G]. Fundus autofluorescence revealed a faint mottled hyper- and hypoautofluorescent pattern in the periphery of the retina [Figure 2H]. In contrast to fundus autofluorescence, multicolor imaging of the peripheral retina detected a vast number of lesions much more visible than in the autofluorescence images [Figure 2E–F] .In multi-color images, when the images from the three different channels were separated to red, green, and blue, we found that the infra-red reflectance photos did not show the lesions, but the blue and green reflectance images highlighted the xerophthalmic lesions similar to the multi-color images [Figure 3].A full-field standard electroretinogram  detected a bilateral reduction in amplitude of rod response and also a smaller reduction in combined rod-cone, oscillatory potential, and cone responses [Figure 4]. According to these findings, we suspected vitamin A deficiency. Serum level of vitamin A was 2 μg/dL  and the diagnosis was confirmed.The patient was treated with intramuscular vitamin A followed by daily administration of oral 50,000 IU of retinol palmitate. The patient did not come back for follow-ups, but upon enquiry, he reported that nyctalopia improved.Nowadays, one of the most common causes of vitamin A deficiency is malabsorption after bariatric surgery, like in the case of the current patient. According to previous reports of xerophthalmic fundus, the most common finding in OCT is the disruption of the outer retinal layers, especially the ellipsoid zone and SDD-like lesions in the peripheral retina.,4,5 Additionally, multicolor imaging was performed and we found that xerophthalmic retinal lesions were much more detectable by fundus multicolor imaging compared to the infrared reflectance photos. Location of the lesions above the RPE may have caused better visibility of lesions in the green and blue channels. To the best of our knowledge, this is the first report of multicolor imaging in the xerophthalmic fundus.Xerophthalmic fundus lesions as presented in this patient are the accumulation of disrupted photoreceptors' outer segments  based on histopathological evaluation.In summary, acquired nyctalopia needs comprehensive fundus examination and paraclinical evaluations, especially multicolor imaging, to have a better detection of the subtle xerophthalmic fundus changes in vitamin A-deficient patients. Early diagnosis and treatment prevent permanent disruption of photoreceptors and subsequent visual impairment.None.The authors declare that they have no conflict of interest.


Cerebellopontine angle tumor (CPA) in pediatrics is rare as compared to adults. We describe a case of pediatric pilocytic astrocytoma presented as a right CPA mass with a concurrent clinical diagnosis of neurofibromatosis type 1 (NF1).A 14-year-old boy with a newly diagnosed hypertension presented with a short history of headache and blurring vision. Neurological examination revealed bilateral papilloedema, partial right third nerve palsy and mild sensorineuronal hearing deficits. Skin examination identified multiple café au lait spots with cutaneous neurofibromas. Preoperative neuroimaging suggested the diagnosis of an extraaxial CPA mass consistent with meningioma, with obstructive hydrocephalus. A left ventriculoperitoneal shunt was inserted and the child was subjected for a suboccipital retrosigmoid approach for tumor resection. The histopathological features, however, were typical for pilocytic astrocytoma.A careful evaluation of the clinical presentation and radiological images of CPA lesions is necessary prior to surgical embarkment. To the best of our knowledge, this case is the first report of pilocytic astrocytoma in the CPA in pediatric with NF1. Cerebellopontine angle (CPA) tumors in pediatrics are rare as compared to adults. The incidence was previously reported at 1%, more frequently benign (> 70%) than malignant and likely to occur on the left side , rather CPA glioma is discussed in the literature as case reports . PilocytA 14-year old boy with a newly diagnosed hypertension presented with a two-week history of right occipital headache that is radiating to the neck associated with blurring of vision. Neurological exams revealed bilateral papilloedema grade II, partial right third nerve palsy and sensorineuronal hearing deficits. The motor, sensory and cerebellar exams were unremarkable. Skin examination identified multiple café au lait spots over the thorax and abdomen with cutaneous neurofibromas. His father was diagnosed with NF1 in childhood. A noncontrast computed tomography (CT) showed a right extraaxial CPA lesion measuring 3.5 cm × 3.5 cm × 2.5 cm with obstructive hydrocephalus. There was a widening of the internal acoustic canal (IAC) (Fig. Intraoperatively, after gentle medial retraction of the cerebellum, we encountered a solid with multiple cystic tumors over the cerebellopontine cistern. Intraoperative monitoring was utilized for trigeminal, facial and lower cranial nerve electromyography, motor and somatosensory evoked potential. The seventh nerve was pushed anterior and superiorly while the eighth cranial nerve was encased by the tumor. The trigeminal and lower cranial nerve was identified and preserved. The tumor was pale to yellowish in color, soft in consistency with moderate vascularity. The tumor center was debulked using a cavitron ultrasonic aspirator until the tumor capsule became mobile. A demarcated arachnoid cleavage was identified with interface between the tumor and cerebellum, middle cerebellar peduncle and part of the pontine region. The tumor abutting the pontomedullary junction was not removed to prevent neurovascular injury.Gross pathologic examination revealed fragments of pale to greyish tumor tissue. Microscopic examination revealed a biphasic appearance: the compact fibrillar area composed of elongated nuclei, bipolar piloid processes and Rosenthal fibers, and the loose microcystic cavity with oval-shaped nuclei. There were scattered eosinophilic granular bodies with observed hyalinised blood vessels. There was no mitosis or endothelial proliferation seen. Immunohistochemical stains were strongly positive for glial fibrillary acidic protein and negative for p53. The Ki67 index is less than 1%. These histopathological features were typical for pilocytic astrocytoma Fig. . His posThe differentiation between extraaxial and exophytic extension of primary brain tumor into the CPA can be appreciated intraoperatively. The tumor located extraaxially has an arachnoid cleavage plane between the tumor and cerebellum, middle cerebellar peduncle, and the pons . Other tChildren with NF1 may develop a benign or malignant intracranial tumors. Tumors related to posterior fossa are cerebellar astrocytoma, medulloblastoma and brainstem gliomas –14. The We presented the first pediatric case of right CPA pilocytic astrocytoma with a concurrent clinical diagnosis of NF1. A careful evaluation of preoperative MRI images of CPA lesions in such patient is necessary and to bear in mind other presumptive pathology prior to surgical embarkment.


Granuloma annulare (GA) is a common benign granulomatous inflammatory disorder of the dermis or subcutis with classic morphologic and histologic presentation. Common clinical subtypes of granuloma annulare include localized, generalized, subcutaneous, perforating, and patch types. A biopsy is critical in cases of granuloma annulare with atypical features. We present a case of a 58-year-old male who presented with an annular scaly erythematous plaque on the right dorsal hand. Shave biopsy demonstrated irregular nests of mildly atypical squamous epithelium present within the superficial dermis, with abundant histiocytes and multinucleated giant cells arranged in palisades peripherally. These findings were consistent with invasive well-differentiated squamous cell carcinoma (SCC) with surrounding granuloma annulare. This case highlights a unique presentation in which clinicopathologic correlation is critical prior to arriving at a correct diagnosis. Granuloma annulare (GA) is a common benign granulomatous inflammatory disorder of the dermis or subcutis with classic morphologic and histologic presentation. We present a case where clinicopathologic correlation led to the diagnosis of invasive squamous cell carcinoma (SCC) within a well-established lesion of GA. To our knowledge, this is the first report of these two diagnoses coexisting as a single lesion in the literature. Morphologic presentation of GA is commonly annular or arcuate, non-scaly, red to brown papules or plaques hypopigmented centrally relative to the periphery. While there are several variants of GA, the localized variant is the most common subtype. The hands, feet, arms, and legs are the sites of predilection in approximately 80% of cases . HistoloDespite GA’s classic presentation, it has been known to mimic other diagnoses. Further, other conditions have been reported to masquerade as GA, including borreliosis, Kaposi sarcoma, and tuberculoid leprosy . Our casA 58-year-old male presented with a new raised skin lesion on the right dorsal hand. He reported that the lesion had grown for a couple of months. He denied any trauma to the area. He denied a history of similar lesions. He denied any systemic symptoms, including fevers, chills, weight loss, and night sweats. He denied a prior personal or family history of skin cancer. On examination, there was a 2.1 × 1.6 cm annular pink plaque with raised borders and central erosion located on the right dorsal hand  with surrounding GA, and the patient was referred for Mohs micrographic surgery (MMS). The patient was cleared by MMS after 1 stage. The preoperative size was 27 × 25 mm, and the postoperative defect was 32 × 35 mm. The defect was partially closed using the purse-string closure technique with the center left open to secondary intent.GA is a granulomatous inflammatory disorder thought to be most common among young adults, especially women . While tDue to its classic morphologic presentation, GA is often a clinical diagnosis. The absence of scale and other secondary features can help assist in the diagnosis of GA . HoweverSCC has been known to arise in chronic wounds, known as a Marjolin ulcer . The oriGA is a benign granulomatous lesion that is typically a clinical diagnosis. The absence of surface change and scale is a crucial finding in differentiating GA from other malignant lesions. A biopsy is critical in cases of GA where morphology is atypical. This case demonstrates a rare presentation of both SCC and GA presenting in a “single” appearing lesion. Importantly, this case highlights a unique presentation for which clinical correlation is imperative prior to arriving at a correct diagnosis and treatment intervention.


Large benign hepatic masses as a rare indication for liver transplantation have been reported less frequently. These liver transplantations are complex, with high intraoperative bleeding, high perioperative complication rates, and high mortality rates due to difficulties in visualization, especially when they have undergone various percutaneous operations or open surgery, resulting in severe perihepatic adhesions. Here is a case report of a patient admitted to our hospital who underwent liver transplantation after suffering from a giant hemangioma in liver transplantation for 10 years and who had received multiple interventional treatments ineffective in the past. Hepatic hemangioma (HH) is the most common benign tumor of the liver. It is usually considered to be a vascular malformation, possessing a vascular nature with a flattened endothelial cell lining, probably related to excessive development or differentiation during embryonic development, and manifesting as well-defined vascular-rich lesions. Most HH originates from the right lobe of the liver, and its pathophysiology has not been clearly elucidated. HH can be divided into subtypes such as cavernous hemangioma, capillary hemangioma, epithelioid hemangioendothelioma, and sclerosing hemangioma according to the amount of fibrous components contained in the tumor, among which hepatic cavernous hemangioma is the most common. Herein, we report a case of a 9-kg giant HH that underwent liver transplantation and evaluate the available literature.in situ whole liver transplantation with a good quality donor liver, and the hypothermic ischemia time, warm ischemia time, and anhepatic phase were 165, 2, and 63 min, respectively, for a total operative time of 385 min. The entire liver and the hemangioma were completely removed , Murphy’s sign (-), normal bowel sounds, and no edema in both lower limbs. Pre-transplantation ancillary examinations: routine blood, coagulation function, liver and kidney function, electrolytes, blood sedimentation, tumor markers, calcitoninogen, and other test results did not show significant abnormalities. Enhanced CT of the upper abdomen: the liver was significantly enlarged, and the lower edge reached the right iliac fossa, suggesting a giant hemangioma of the liver . The cli removed , and a n removed . PostopeWith the gradual increase in the understanding of HH and the rapid development of imaging techniques, the detection rate of HH has been increasing, with a reported incidence of 0.4–20% and a male-to-female ratio of approximately 1:5  that hemHH is usually asymptomatic and is detected only during physical examination, dissection, or autopsy. Approximately, 61% of patients have small, solitary lesions that grow slowly and have a long course, and patients may have no significant abnormalities in liver function . GeneralThe diagnosis of HH currently relies on imaging, and the unique features of HH imaging are the presence of peripheral nodal enhancement and progressive centripetal filling, with ultrasound (US), computed tomography (CT), and magnetic resonance imaging (MRI) being the most commonly used examinations. Among them, US is the preferred method of examination and presents as a well-defined, homogeneous hyperechoic mass that can be deformed under pressure, and larger hemangiomas show mixed echogenicity, with internal echoes still predominantly hyperechoic. Although US is more sensitive, its specificity is only 60.3% because it only highlights blood flow and intra-arterial portal shunts in HH, and some malignant liver lesions  may also produce similar acoustic patterns , 8. HH in = 4), persistent abdominal distension, abdominal pain (n = 3), thrombosis due to compression of the inferior vena cava (n = 2), and bleeding from ruptured mass (n = 1), as detailed in HH is asymptomatic in most cases and has no malignant tendency, so, in principle, no treatment is needed, and follow-up observation is the main focus. Treatment should be limited to patients with complications, such as symptoms, persistent enlargement of the mass, compression of adjacent organs , hemangioma rupture, and bleeding or Kasabach-Merritt syndrome (KMS), which is an internationally accepted concept, and its treatment includes radiofrequency ablation, hepatic artery interventional embolization, surgical resection, and liver transplantation . AlthougAlthough surgical resection is still the most effective treatment for hepatic hemangioma, hepatectomy requires consideration of the residual function of the liver, the condition of the liver, the size and the number of tumors, and the location of the tumor. Considering that the tumor occupies almost the whole liver and the liver residue is insufficient after resection, which may lead to small liver syndrome, plus the fact that the patient’s interventions have been ineffective for many times since the patient’s illness, and the symptoms have been worsening, with the possibility of rupture and compression symptoms, which are indicative of liver transplantation, liver resection was not considered, and the patient was placed on the liver transplantation waiting list.It is noteworthy that, in this case, due to the large size of the diseased liver, it was difficult to reveal it. In order to fully expose the operative field, the surgical incision should be large enough, so we chose a “herringbone” incision in the upper abdomen, which was about a 40-cm long, and then entered the abdominal cavity layer by layer. The lower edge of the liver reached the pelvic cavity. The huge liver was clearly adherent to the surrounding intestinal tract, and a clearly edematous gastrointestinal tract could also be seen, which posed considerable difficulties in continuing the operation. In this case, the first hepatic hilar and inferior hepatic vena cava were preemptively blocked to avoid hemorrhage when the liver was freed. By blocking the blood flow to the liver, the size of the HH was gradually reduced, thus allowing full exposure of the operative field. Meanwhile, in addition to the important role of surgical technique and experience in limiting blood loss, intraoperative venous bleeding can be reduced by lowering central venous pressure (CVP) and portal vein pressure, thereby reducing collateral vessel filling, which, in turn, helps to reduce intraoperative venous bleeding. Methods to reduce CVP include fluid volume restriction, venous dissection, use of diuretics, such as mannitol, low tidal volume ventilation, and avoidance of high positive end-expiratory pressure ventilation. The potential benefits of lowering CVP should outweigh the risk of inadequate organ perfusion and renal injury to the patient. The need to meet tissue oxygen requirements while reducing CVP can be assessed by measuring mixed venous oxygen saturation, arterial blood lactate, and other parameters. Intraoperative bleeding in liver transplantation is closely related to the severity of the disease, coagulation, and surgical proficiency. The first thing to note is the patient’s coagulation status, while changes in prothrombin time and activated partial thromboplastin time have limited significance in determining coagulation function intraoperatively. Intraoperative thromboelastography (TEG), which dynamically reflects changes in blood coagulation status, can be used to monitor the patient’s coagulation profile to assess coagulation function, guide the use of medications and evaluate their effects, avoid massive blood transfusions due to disorders in coagulation mechanisms, and reasonably transfuse blood products. These include suspended red blood cells, fresh frozen plasma and platelets. In addition, intraoperative blood retrieval transfusions can be performed by collecting the recipient’s own blood or retrieving blood lost in the intraoperative field to meet his or her own perioperative blood supply. Intraoperative recovered autologous transfusion uses a negative pressure suction device to collect blood lost during surgery, and then returns it to the patient after filtering, separation, washing, purification, and concentration. Autologous blood transfusion can not only save limited blood resources and reduce the economic burden of patients but also avoid a series of adverse reactions and potential infectious diseases caused by massive transfusion of allogeneic blood during liver transplantation.After complete resection of the diseased liver, we were faced with the challenge that the donor liver was relatively small compared to the diseased liver, and the length of vessels and bile ducts required for implantation of the new liver had to be adequate for transplantation. The duodenal ligament can be dissected as close to the liver as possible in order to preserve sufficient length of vessels and bile ducts, and, in view of the large difference in size between the diseased liver and the donor liver, the liver may be displaced after implantation. To prevent this, the perihepatic ligament was further sutured and secured in the anterior abdomen during surgery, and intraoperative ultrasound was used to determine the patency of the vessels and bile ducts. In addition, we believe that resection of a large diseased liver is often difficult, and there are a number of uncertainties that can lead to prolonged operative times. Firstly, the prolonged duration of the operation to remove the diseased liver leads to prolonged hypothermic ischemia time of the donor liver. Secondly, uncontrollable hemorrhage during resection of the diseased liver requires rapid removal of the diseased liver, which can lead to a prolonged anhepatic phase if the donor liver has not yet been repaired. Thirdly, dissection of the diseased liver is difficult, and the operation takes longer. Therefore, when facing this type of surgery, not only do we need to be familiar with the anatomical location and cooperate carefully intraoperatively, but, also, the donor liver acquisition, diseased liver resection, and recipient surgery teams should communicate in a timely manner and actively improve the preoperative preparation so that the donor and recipient surgery can be more closely connected to facilitate successful surgery.Although the best treatment for giant HH is not yet uniform, liver transplantation has been shown to be a safe and effective treatment in cases where conservative treatment is ineffective and surgical resection is not possible.The original contributions presented in this study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.YZ was responsible for formulating writing ideas and writing the manuscript. X-PL, Y-YH, and J-CJ were responsible for collecting information. L-JZ was responsible for revising the manuscript and guiding the writing of the manuscript. All authors contributed to the article and approved the submitted version.


Cerebral amyloid angiopathy (CAA) is the most common cause for lobar haemorrhages. The prevalence of CAA is believed to be app. 30% in non-demented elderly patients and increases with age . CAA is Recently, the imaging signs “finger like projections” (FLP) and subarachnoid haemorrhage (SAH) adjacent to the main haemorrhage have been suggested as additional markers of CAA .While MRI is desirable to advance the diagnosis of CAA non-invasively, in some patients, MRI is not feasible due to contraindications or logistics constraints. Moreover, plain CT remains the first-line imaging modality in the acute stage.We present a retrospectively compiled case series of patients with lobar haemorrhages in whom an MRI was not performed but many imaging features hinted towards the diagnosis of cerebral amyloid angiopathy on plain CT.An 81-year-old patient presented at a three months interval first with left and then with bilateral sensorimotor symptoms. CT demonstrates a left parietal lobar haemorrhage and bilateral frontal lobar haemorrhages with extension into the subarachnoid space involving the left sulcus of the corpus callosum and the right central sulcus Fig. -C. No suA 68-year-old male patient presented with reduced level of consciousness and severe left sided hemiparesis. Two months later, he again presented with reduced level of consciousness and severe left sided hemiparesis. On both instances, CT demonstrates a large right sided lobar haemorrhage with SAH and FLP Fig. -G. On boAn 82-year-old male patient presented with headaches and severe non-fluent aphasia. CT demonstrated a left temporal lobar haemorrhage with subarachnoid and subdural extension. FLP could be demonstrated Fig. -J. CraniIn all cases, specimens obtained from hematoma evacuation underwent neuropathological assessment including immunohistochemistry staining which demonstrated severe Aβ-amyloid angiopathy Fig. .Fig. 2TiThe modified Boston criteria require evidence for multiple haemorrhagic lesions demonstrated either clinically, on CT or MR for the diagnostic category “probable cerebral amyloid angiopathy” . The casThese cases remind us that plain CT holds a wealth of information if MRI is not available.


Toothbrush ingestion is a rare phenomenon. It is usually found in psychiatric, elderly and mentally disabled patients. Foreign bodies usually pass spontaneously and uneventfully through the gastrointestinal tract. Nevertheless, larger objects may require early intervention to avoid complications. This report describes the course of treatment for a 25-year-old woman with an accidentally ingested toothbrush. Foreign body ingestion is a typical emergency case . Most smA 25-year-old female presented to the accident and emergency department with a history of accidentally ingested toothbrushes. The patient had a history of anorexia nervosa with multiple episodes of induced emesis. On presentation 3 hours post-ingestion, she was asymptomatic and vitally stable, with no physical abdominal findings. X-ray abdomen was ordered, which confirmed the presence of the toothbrush at the left upper quadrant . The patIngestion of a foreign body is commonly seen in children and adults with mental impairment, psychiatric illnesses and alcoholism as well as in elderly individuals who wear prosthetic dental materials . HoweverIn most cases, the preferred extraction tool was a basket or polypectomy snare. However, in our case report, we used a gastric balloon grasper to remove the toothbrush . This haToothbrush extraction is a challenging case that requires an experienced and skilled gastroscopist. Early extraction is crucial to avoid morbidity and mortality. A failed endoscopic attempt may require laparoscopy or laparotomy to extract it successfully; thus, it is advisable to have a surgeon in the operating room.None declared.None.All data are available in the article.


Parastomal hernia (PH) is an abnormal herniation of tissue or intra-abdominal organ through the fascial defect created at the ostomy site. It is a common complication of stoma creation and usually contains bowel, intra-abdominal fat, or omentum. Herniation of a fixed organ like the stomach is very rare and can lead to significant morbidity and mortality. Here, we present a case of an 83-year-old female with a history of sigmoidectomy and subsequent development of parastomal hernia who presented with abdominal pain, nausea, and vomiting and was found to have stomach herniation into the parastomal hernia sac. She was managed conservatively with intravenous (IV) fluids, electrolyte replacement, and decompression with a nasogastric (NG) tube. In this article, we have discussed the incidence, clinical presentation, diagnosis, and management of gastric involvement in the parastomal hernia that can help clinicians identify and treat it early at the time of presentation. Although there have been recent advancements in surgical practices and techniques, the incidence of parastomal hernia (PH), along with its complications, has been on the rise . A parasAn 83-year-old female patient, with a history of sigmoidectomy, presented with two weeks of abdominal pain. The patient previously had a large, highly vascular 2 cm polyp in the recto-sigmoid region that lead to a sigmoidectomy with an end colostomy approximately two years prior to admission. After surgery, she developed a parastomal hernia and was being followed by surgery in the outpatient setting. The abdominal pain was gradual in onset, non-radiating, located around the site of hernia, 5/10 in intensity, and was associated with abdominal distention, nausea, vomiting, and large volume output from her colostomy. At the time of admission, vitals were as follows: temperature 97.4 F, pulse 91/minutes, blood pressure 186/111 mmHg, respiratory rate 18/minutes, and saturation 97% on room air. On examination, the patient was in mild distress. An abdominal examination demonstrated a colostomy bag in the left lower quadrant with an underlying large, soft, fluctuant mass without any discharge from the ostomy bag. No tenderness to palpation, guarding, rebound tenderness, or rigidity was noted. Initial laboratory studies were significant for white cell count 7.8, hemoglobin/hematocrit 12.1/36.8, platelet count 257, sodium 145, potassium 3.2, chloride 101, bicarbonate 30, blood urea nitrogen 51, and creatinine 3.73. A computerized tomography (CT) abdomen and pelvis showed a parastomal hernia containing a portion of the distal stomach and signs of associated gastric outlet obstruction with severe gastric dilation Figure -2D. GeneParastomal hernia (PH) is an abnormal herniation of tissue or an intra-abdominal organ through the fascial defect formed at the ostomy site . It is aGPH is extremely uncommon, in part due to the stomach’s unique intra-abdominal and anatomically secure location. It is surrounded by the diaphragm, duodenum, esophagus, liver, spleen, and transverse colon and is reinforced by multiple ligaments that provide support, integrity and prevent its herniation -3. It haThere are numerous risk factors for GPH. These can be categorized as patient-related, technical, and surgical risk factors. The patient-related risks include female gender, malignancy, infection, chronic obstructive pulmonary disease (COPD) , emergent procedures, advanced age, obesity, malnutrition, use of immunosuppressive drugs, and diabetes mellitus. The technical factors include the size and type of the stoma, with an increased incidence being stated in end stoma colostomies as compared to loop stomas and ileostomies. There was also a higher incidence in stomas >25 mm as compared to small ones ,2,6,7,10Although mostly asymptomatic, PH carries an increased risk of becoming incarcerated or strangulated leading to perforation, gangrene, or obstruction of the organ involved. The clinical manifestation of GPH depends on the extent of gastric involvement. Most studies have shown either partial or complete gastric outlet obstruction as the presenting complaint, with symptoms ranging from abdominal pain, nausea, vomiting to strangulation and perforation -3. In thGPH is usually diagnosed with a good history and physical examination. Imaging studies, including plain films with contrast or abdominal computerized tomography (CT) scans, are often utilized to identify the severity, location, contents, and complications of GPH, including perforation. Some case studies also mentioned the use of imaging studies, including fluoroscopic examination of the upper gastrointestinal tract (GI) tract to diagnose gastric parastomal hernia ,3. As inThe management of GPH depends on its severity. With mild symptoms or patients with partial gastric outlet obstruction, treatments usually include nasogastric (NG) decompression, serial abdominal exams, and stoma care using an ostomy belt or abdominal binder. In more severe cases, initial management should include NG decompression followed by surgery. Indications for surgery include failure of conservative management, persistent symptoms including severe pain, distention, nausea, vomiting, irreducible bulge, obstipation, or if the patient manifests life-threatening conditions including gastric perforation, incarcerated or strangulated hernia, gastric emphysema, or septicemia. Surgical management includes stoma relocation, ostomy reversal with mesh repair for better tissue repair. Many studies have also shown a reduced incidence of parastomal hernia with prophylactic mesh placement at the time of first stoma creation ,3,5-7,10This case highlights the importance of gastric parastomal hernia (GPH), which is a very rare phenomenon, mostly seen in elderly females in their seventh to eighth decade of life who had end colostomies created within 10 years. The risk of GPH increases with female gender, malignancy, chronic obstructive pulmonary disease, advanced age and end colostomies, most of which were also present in our patient. Clinical presentation of GPH can range from being asymptomatic to causing vomiting, nausea, abdominal pain, strangulation, incarceration, and perforation. They are usually diagnosed with a good history and physical exam along with the computerized tomography (CT) scan and an upper endoscopy to rule out intraluminal causes of gastric outlet obstruction. Treatment of GPH includes conservative management with nasogastric (NG) decompression, serial abdominal exams, and stoma care, followed by surgery for persistent and worsening symptoms.


Pelvic pain in reproductive age often represents a diagnostic challenge due to the variety of potential causes characterized by overlapping clinical symptoms, including gynecological and other disorders . It is also necessary to determine if there is a possibility of pregnancy to rule out any related complications, such as ectopic pregnancy. Although ultrasound (US), computed tomography (CT), and magnetic resonance imaging (MRI) are strongly integrated, the choice of which is the ideal diagnostic tool should be guided both by clinical suspicion  and by the risk ratio–benefit , too. The didactic objective proposed by this review consists in the diagnosis of the cause and differential of pelvic pain in reproductive age by describing and critically analyzing the US diagnostic clues of the most frequent adnexal, uterine, and vascular causes. Pelvic pain in reproductive age often represents a diagnostic challenge due to the variety of potential causes characterized by overlapping clinical symptoms, including gynecological and other disorders . It is also necessary to determine if there is a possibility of pregnancy to rule out any related complications, such as ectopic pregnancy . SeveralPelvic US is considered the ideal investigative tool to use at the beginning of the diagnostic and evaluation process for suspected gynaecological disorders in patients of all ages. US has the advantages of being widely available, low cost, and free from ionising radiation. Additionally, US is often sufficient to diagnose uterine, ovarian, and adnexal pathologies . US is aHowever, the characteristics of pelvic pain must be strongly considered before any instrumental diagnostic approach. In this regard, Adenxal torsion is the partial or complete rotation of the adnexum on its vascular peduncle resulting in congestion and oedema, due to compromised venous and lymphatic drainage, and subsequent ischaemia and necrosis, due to compromise of the arterial blood supply ,4. AlthoIt can affect the adnexum in its entirety involving both the ovary and the fallopian tube . The torSince it is defined as a surgical emergency, an early differential diagnosis including other genitourinary and gastrointestinal conditions is essential to preserve the ovaries. The clinical symptoms are characterized by acute and sudden pain, sometimes associated with nausea and vomiting with the finding of a palpable pelvic mass and signs of peritonitis ,3. The iThe ovary may be found in an abnormal position, close to the midline of the uterus, higher or lower in the pouch of Douglas, or displaced to the contralateral ovarian space ,4,5. TheThe twisted and congested peduncle may appear as an indistinct adnexal mass adjacent to the twisted ovary. The colour Doppler examination allows for the assessment of the degree of torsion, the time elapsed from the beginning of the disease and therefore the degree of vascular compromise ,2,3,4,5.However, the presence of flow does not exclude torsion, both due to the two-fold perfusion of the ovary by the ovarian and uterine arteries, despite the initial loss of venous flow, and because the torsion may be intermittent or incomplete. However, Doppler spectral waveform analysis can increase the sensitivity of the torsion diagnosis because an arterial waveform with diastolic flow inversion, indicating high resistance, may suggest the presence of torsion  1,2,3,4,43,4,5.Ovarian cysts are growths that develop in the ovaries during follicular maturation and are defined as functional cysts as they represent a physiological phenomenon related to ovarian function ,6. FunctThe fibrin bundles produce a network of precise linear echoes and are distinguishable from the real septa because they are numerous, thin, and dispersed, with an irregular structure and are not vascularised on colour Doppler US or contrast-enhanced US (CEUS)  1]..1].In addition, unlike a true septum, they do not extend from one wall of the cyst to the other. The lack of flow detected by the colour Doppler imaging is an important feature, suggesting the presence of a mural thrombus rather than a tumour nodule. However, low levels of flow in solid tissue may not be detected by colour Doppler US, whereas contrast CEUS may optimise the diagnosis when clinical suspicion is present  1,6,7,8,7,86,7,8Pelvic inflammatory disease (PID) is an infection of the female genital tract caused by the ascent of microorganisms from the vagina to the uterus, fallopian tubes, and ovaries. Common causes are Neisseria gonorrhoeae and Chlamydia trachomatis, which are sexually transmitted ,9. OtherNo specific international data are available for PID incidence worldwide. However, among 1171 sexually experienced reproductive-aged women in the 2013–2014 National Health and Nutrition Education Survey (NHANES), the prevalence of self-reported lifetime PID was 4.4% .PID can be acute, chronic, or subclinical and is often underdiagnosed. Symptoms include lower abdominal pain, fever, mucopurulent discharge, and abnormal uterine bleeding during or after menstruation. Untreated PID can lead to chronic pelvic pain, infertility, ectopic pregnancy, and intra-abdominal infections. The diagnosis is made primarily on clinical suspicion . In thisOn US, infected fallopian tubes may show thickened and hyperaemic walls, which are dilated in the presence of pyosalpinx with occluded ovarian fimbria, echogenic intraluminal sediment, and stratified echoes that signal the presence of exudate  9]..9].The inflamed fallopian tube appears adjacent or adherent to the ovary with the formation, in the most advanced cases, of an ovarian abscess represented on US by an inflammatory mass which engulfs the ovary and the tube, making the ovary no longer distinguishable  1,9].,9.1,9].Rupture of tubo-ovarian abscess can result in septic shock  1,9].,9.1,9].Endometritis is inflammation of the endometrial lining of the uterus that could also involve the myometrium and, occasionally, the parametrium too, and clinically manifest with fever, chills, lower abdominal pain, and foul-smelling lochia or PID related symptoms ,13. It cOn US, inflammation of the endometrium can show a thickened endometrium with an irregular profile and the presence of more or less echogenic fluid or pus in the uterine cavity (pyometra)  Figure  14]..14].Endometriosis is a chronic inflammatory condition caused by the abnormal presence of endometrial tissue at sites other than the physiological endometrium. It affects up to 10% of women of reproductive age, is often asymptomatic and, when symptomatic, it manifests with chronic pelvic pain, dysmenorrhea, dyspareunia, and abnormal uterine bleeding ,16,17,18Endometriomas form part of the differential diagnosis alongside various ovarian cystic formations such as luteal, haemorrhagic, and dermoid cysts particularly. US guides the differential diagnosis, since, in the case of haemorrhagic cysts, monitoring in the different phases of the menstrual cycle allows for the observation of spontaneous resolution of the cysts, unlike endometriomas, which usually persist. In the presence of a non-definitive US examination or suspected ovarian cancer, an MRI scan is recommended ,16,17,18However, the characteristics detected are variable. The edges may be serrated and infiltrated by adjacent soft tissues. Most disease deposits show vascularisation on colour Doppler. The differential diagnosis includes abdominal wall masses, such as desmoid tumour, metastasis, lymphoma, melanoma, haematoma, suture granuloma, or incisional hernia; however, the location of the mass adjacent to a scar and a history of cyclic pain consistent with menstrual pain guide the diagnosis  1,15,1616,17,18.Peritoneal inclusion cysts (PICs) are generally benign mesothelial lesions with an estimate incidence of approximately 3–5% of the peritoneal mesotheliomas containing peritoneal fluid that occur in women of childbearing age, often following invasive pelvic surgery, infection, or cancer .This condition is most commonly described in women of reproductive age with a ratio of 4–5:1 female to male, with very few cases reported in females over 30 years of age .Intra-abdominal inflammation with reactive mesothelial proliferation and impaired absorption of peritoneal fluid can lead to its formation ,21.PICs are irregularly shaped, multiloculated adnexal cystic masses with angled margins, lacking a wall of their own and moulding themselves around the surrounding pelvic organs within the peritoneal cavity. A common clinical presentation is a progressive lower-abdominal and pelvic pain or a palpable mass ,21. Due imaging features of peritoneal inclusion cysts reflect their pathogenesis and allow for their differential diagnosis from an ovarian cystic mass .,41.40,41In this regard, it appears critical to have a rigorous methodological US approach of pelvic and retroperitoneal veins combining TSA–US and TVS–US with dynamic colour-Doppler Valsalva maneuvers performed in the patient’s supine, half-sitting (with trunk raised to 45°), and half-standing positions ,41. TVS–Finally, duplex-ultrasonography of the veins of lower extremities is also a necessary part of the imaging protocol for improved evaluation of pelvic congestion syndrome, especially in the presence of atypical varicose veins .Gonadal vein thrombosis is a condition that can occur in postpartum women or those undergoing pelvic surgery, with a referred incidence of about 0.18% of the general population ,44. AffeCT or MRI, both with contrast medium, should be carried out in the event of an uncertain diagnosis and to assess the extent of thrombosis . FurtherPelvic US is the ideal investigative tool to adopt as first line examination for suspected gynaecological disorders in patients of all ages. As pictured, US allows for an accurate diagnosis of ovarian torsion, hemorrhagic ovarian cyst, endometriosis, pelvic inflammatory disease, complications related to leiomyomas, and vascular anomalies first by placing them in the differential diagnosis and between other non-gynecological causes of pelvic pain such as gastrointestinal and genitourinary disorders. The recognition of such disorders can make timely diagnosis possible and avoid further imaging tests.


Ovarian hemangiomas are generally asymptomatic. However, associated stromal luteinization, reported in some cases, may lead to the development of hyperandrogenic syndrome. We report the case of a 61-year-old female referred to the endocrinology outpatient clinic for hirsutism, hair loss, and deepening of the voice tone. The investigation showed high serum testosterone and normal dehydroepiandrosterone sulfate (DHEAS). Normal ovaries were observed in the initial transvaginal ultrasound, but an abdominal-pelvic nuclear magnetic resonance imaging (MRI) identified a solid mass in the right ovary. The patient underwent surgery, and pathological examination revealed a capillary-type ovarian hemangioma with stromal luteinization. After the surgery, clinical and analytical response was very favorable. During menopause, ovaries maintain some residual hormonal activity. There is an imbalance between estrogens and circulating androgens, in favor of the latter, amplified by the decrease in the concentration of sex hormone-binding globulin (SHBG), which can lead to the appearance of clinical signs of hyperandrogenism ,2. HowevOvarian hemangiomas are benign, rare, and usually small-sized tumors, so their detection can be a challenge. Stromal luteinization, reported in some cases in the literature, is an uncommon process and is responsible for the secretion of androgens, which can lead to hyperandrogenic syndrome. Its pathogenesis is unknown and controversial ,4.2; a low tone of voice; hirsutism in the face, chest, and abdominal region ; enhanced muscle development at the scapular level and deltoid region; and rarefaction of the scalp in the frontoparietal region. There was no evidence of acanthosis nigricans or cushingoid phenotype. Her blood pressure was 126/72 mmHg. Her abdominal examination did not reveal masses or organomegaly. Gynecological inspection showed clitoromegaly. Blood count, fasting blood glucose, ionogram, and renal and liver function tests were normal. Her lipid profile was deranged. The hormonal study revealed very high total and free serum testosterone, favoring an androgen-secreting tumor  was performed, which identified a solid mass measuring 37 × 20 mm, relatively well defined in the right ovary Figure , and 13 The ultrasound was repeated, but it was not possible to visualize the ovaries endovaginally. However, the abdominal approach allowed the identification of a solid mass in the right ovary. The left ovary was apparently normal. There was no evidence of ascites. Given the high value of 17-alpha-OH-progesterone (17-OHP), a genetic study was carried out that showed a variant in heterozygosity in the CYP21A2 gene, variant c.844G> T , exon 7, which excluded the diagnosis of congenital adrenal hyperplasia. Given the adrenal incidentalomas, she underwent screening for pheochromocytoma and Cushing’s syndrome: urinary metanephrines were normal, and 1 mg dexamethasone suppression test = 2, pointing to a possible autonomous secretion of cortisol. Spironolactone was started at 100 mg, with a progressive dose increase up to 200 mg/day, and finasteride 5 mg, without signs of clinical improvement. Considering the suspicion of virilizing ovarian tumor, the patient underwent a total hysterectomy and laparoscopy bilateral annexectomy, which revealed isolated ovarian involvement. Peritoneal lavage was negative for neoplastic cells. The procedure was uneventful, and the patient was discharged on the fourth day after surgery. Five months after the surgery, the androgens and lipids returned to normal , with Leydig cell morphology and others with luteinized aspect, without cytological atypia. CA 125 was negative. The final histopathological diagnosis was anastomosing ovarian hemangioma with stromal luteinization  (Figure No pathological changes were found in the left ovary, fallopian tubes, and myometrium. Currently, the patient is asymptomatic, and no additional treatments were necessary.The etiological diagnosis of hyperandrogenism can be a real challenge. During menopause, in the presence of virilization signs, the hypothesis of an androgen-secreting tumor should be considered ,6. The iOvarian hemangiomas associated with stromal luteinization, reported in some cases, may give rise to the development of hyperandrogenic syndrome ,9. In thThe reappearance of vasomotor disorders after surgery seems to mimic what happens when menopause begins, which is explained by the resolution of hyperandrogenism and consequently the drop in estrogens by the aromatization process. The definitive diagnosis is given by histopathological evaluation and immunohistochemical analysis ,8. A corDuring menopause, the appearance of signs of virilization should prompt the investigation of virilizing ovarian tumors. Although unusual, ovarian hemangioma with stromal luteinization should be considered in the differential diagnosis of hyperandrogenism. A high index of suspicion is warranted because androgen-secreting ovarian tumors may be missed because of their small size.


Vulvar masses in children are an unusual finding but their differential diagnosis is extensive. In case of solid masses, rhabdomyosarcoma (RMS) must always be considered due to the fact that it is the most common tumor in external genitals during childhood. However, RMS has a radiological appearance very similar to juvenile xanthogranuloma (JXG). We present a 16-month-old girl with a 2 cm solid mass on her left labia majora, with four overlying cutaneous papules. After imaging tests, an excisional biopsy was programmed due to high malignancy suspicion. Histopathology of the mass and one of the papules was diagnostic for JXG. After a 12-month follow-up, the patient shows no signs of relapse or complication. Deep JXG is an uncommon entity in childhood and exceptional in the genital area. Therefore, it must be included in the differential diagnosis of a solid vulvar mass, especially if accompanying yellowish xanthomatous cutaneous lesions are present. External genitalia masses in girls are rare. Differential diagnosis is challenging as it is very extensive and not of common knowledge. There are four main etiological groupsA 16-month-old girl with a 2.5-month history of an enlarging vulvar mass on her labia majora was referred to our center by her pediatrician. Family members reported significant growth in the last 24 hours. Otherwise, the patient did not show fever or other symptoms. The mass was neither painful, fluctuant nor discharged purulent material at compression. Examination revealed a hard, well-defined, nonerythematous, nonfluctuating tumor on her left labium majus as well as bilateral inguinal lymphadenopathies. Four erythematous papules were seen on the overlying skin .A perineal ultrasound scan described a poorly defined 28 × 17 × 24 mm cystic image with heterogeneous content associated with perilesional edema .Contrast-enhanced magnetic resonance imaging (MRI) was performed . It shoGiven the suspected malignancy, an open biopsy was scheduled. In the lithotomy position, a left parasagittal incision was made on the left labium majus, revealing a 2.5 cm encapsulated tumor attached to bone, with a cranial extension toward the puborectalis muscle. Due to the good delimitation of the lesion and the absence of macroscopic infiltration, an excisional biopsy was performed. The cutaneous satellite lesions were biopsied at the same procedure. The patient was discharged on postoperative day 7 with a mild surgical wound hematoma in resolution.Histopathological examination  showed After having ruled malignancy out, the patient did not receive any adjuvant treatment. She was referred to the ophthalmology clinic to rule out ocular involvement, which was absent. An abdominal ultrasound was performed without abnormal findings. She is currently followed at the outpatient pediatric oncology and pediatric surgery clinics, without any complications or signs of recurrence after a 12-month follow-up.Vulvar tumors, although infrequent, include a very wide, and in many cases unknown, differential diagnosis.Regarding benign masses, they are mostly mesenchymatous: lipoma,Regarding malignant tumors, the most common lower genital neoplasm in children is RMS.JXG is not usually included the differential diagnosis of vulvar masses. This entity, although unusual, is the most frequent non-Langerhans histiocytosis. It is characterized by single or multiple papules or nodules, initially pink but later turning yellowish. Although most of them are cutaneous, there are subcutaneous and visceral variants. They are typically found in infants and young children, most of them appearing during the first year of life. Its most frequent locations are head, neck, and trunk. Extracutaneous involvement is rare (4% of cases), eye being the most commonly affected organ. Their natural tendency is toward long-term regression; therefore, they usually do not require surgery and it is reserved for atypical cases.Being the upper body its most common location, genital lesions do not usually raise suspicion of this entity. There are very few published articles reporting this location. After an exhaustive bibliographic research, we found a total of 12 cases in children: three patients with lesions on the penis,i-M1P, anti-factor XIIIa, vimentin, and anti-CD4 but negative for CD-1a and S-100.Our patient presented with a short-time history of a vulvar mass without the typical macroscopic characteristics of a JXG, so this diagnosis was not considered at first. The initial diagnostic test for a vulvar mass is a soft tissue ultrasound. Given the impossibility of ruling out a RMS, we performed an MRI to characterize the mass, which, in this case, showed malignancy data. RMS MRI image is similar to that of the JXG, both lesions are hypointense on T1-weighted images and hyperintense on T2, being invasion of other tissues the key sign to distinguish them.After appropriate histological diagnosis, extracutaneous involvement must be ruled out.JXG is a benign pathology that has a tendency toward disappearance over a period of 3 to 6 years. However, its heterogeneity in morphological aspect and evolution, as well as its low prevalence, makes its diagnosis challenging. In many occasions, surgical resection ends up being required to procure histological diagnosis, as it was in our patient's case. Surgical treatment in JXG, although not essential, implies definitive resolution of the disease.Vulvar masses in childhood are infrequent and include a very wide differential diagnosis, from congenital lesions, hormonal disruptions to benign or malignant tumors.Deep JXG is an infrequent entity. Its genital location is exceptional, with only 12 cases described in the scientific literature.The radiological image of the RMS is similar to that of the JXG, so in cases of diagnostic doubt, a biopsy of the mass is essential, since the histological diagnosis is definitive. Although JXG is a self-resolving pathology, diagnostic doubts may force surgical excision, which is also curative.


Subarachnoid hemorrhage (SAH) is a rare manifestation of brain tumors, being even rarer in vestibular schwannomas. We report the second case of a posterior circulation aneurysm close to the tumor capsule, responsible for SAH as an initial manifestation of a vestibular schwannoma. A 67-year-old female was admitted to the Emergency Department with sudden onset of nausea and headache. A diagnosis of a SAH and a tumor of the right cerebellopontine angle was made. An angiography showed an aneurysm in the dependency of the right anterior inferior cerebellar artery (AICA), juxtaposed to the tumor capsule. The patient underwent surgery, the tumor was removed, and the aneurysm was treated. This case highlights that SAH in patients with vestibular schwannoma may originate from a contact aneurysm. Although exceedingly rare, surgeons should consider this scenario in vestibular schwannoma presenting with SAH, and angiography is important for its diagnosis. Subarachnoid hemorrhage (SAH) as the initial presentation of a tumor is an uncommon event. The tumors that bleed most often are astrocytomas, oligodendrogliomas, metastatic tumors, meningiomas, choroid plexus papillomas, and hemangioblastomas ,2.The vestibular schwannoma is a benign tumor, which arises from Schwan cells usually in the vestibular division of the VIII cranial nerve. Its incidence rounds 11-13 cases per million every year and comprises about 6% to 10% of intracranial tumors . The comCases of SAH in patients with vestibular schwannomas and concomitant aneurysms have been previously reported, the majority unruptured and in the anterior circulation ,6. To ouA 67-year-old woman, with past a medical history of stage IIIC breast cancer, stage 5 chronic kidney disease of undetermined etiology, and hypertension, was admitted to the Emergency Department with sudden onset of nausea and intense headache. On physical examination, she was agitated and disoriented, without focal neurological deficits. A cranial unenhanced computed tomography (CT) was performed revealing a spontaneous SAH in the basal and suprasellar cisterns, as well as sylviSylvianures and convexity sulci. Additionally, an expansive cystic lesion in the right cerebellopontine angle plus active supratentorial hydrocephalus were found Figures -1D.In order to understand the bleeding source, a cranial CT angiography was performed, which did not reveal aneurysms or vascular malformations. While in the Emergency Department, the patient’s level of consciousness worsened, and an emergent external ventricular drain (EVD) was placed. The patient recovered and was admitted to the neurocritical care unit. On the following day, she underwent a cerebral angiography with catheterization of the right and left internal carotid arteries and the left vertebral artery, which ruled out aneurysms or vascular malformations. A cranial magnetic resonance imaging (MRI) was also performed to characterize the right cerebellopontine lesion, confirming an apparently cystic lesion with a small solid component measuring 3.7 cm in diameter, whose diagnostic hypotheses were metastases, meningioma or cystic hemangioblastoma Figures -2C.On the sixth day of hospitalization, the patient externalized the EVD with the need for replacement. A postoperative CT scan confirmed correct EVD positioning but showed increased lesion density and additional blood in basal cisterns, reflecting possible re-bleeding Figures , 3B.Thus, a new angiography was performed, this time with the catheterization of the four vessels, revealing a small distal AICA pseudoaneurysm Figures , 4B.It also showed mild to moderate vasospasm in posterior circulation. On the 12th day after admission, the patient underwent a right retrosigmoid craniotomy to excise the expansive lesion. Unexpectedly, the tumor was not cystic as believed based on preoperative imaging exams, but solid, with clots and liquefied hematic content reflecting previous bleeding. The lesion was extensively adherent to the VII cranial nerve Figures -5D.In the final phase of tumor removal, a small aneurysm was identified in the right AICA, in relation to that seen on angiography. It displayed bleeding during gentle manipulation before being excluded with bipolar coagulation. The remaining AICA was preserved, without noticeable infarct areas. Postoperative cranial MRI showed complete tumor removal Figures -6C.The histological result was compatible with a schwannoma. On the 27th day of hospitalization, due to the maintenance of hydrocephalus, a ventriculo-peritoneal shunt was placed. The patient had a favorable clinic evolution, with surgical sequelae of right facial peripheral palsy, with no other neurological deficits.Tumor hemorrhage comprises 1%-11% of the causes of intracranial hemorrhage and may manifest as SAH, intratumoral hemorrhage with SAH, or intraparenchymal hemorrhage around the tumor ,8. ThereThere is also mention of a small number of cases of vestibular schwannomas where unruptured aneurysms of the anterior circulation were concomitantly found ,6. HowevIn patients with vestibular schwannoma who present with concomitant bleeding, tumor hemorrhage should not be assumed as the sole hypothesis. Especially if there is an associated SAH, the patient should be investigated further. In this report, the key to the diagnosis was the high rate of suspicion, which led us to repeat the angiography, leading to the successful treatment of the patient. Thus, in a patient with a vestibular schwannoma presenting with SAH, the presence of an aneurysm near the tumor should be considered a possible cause of bleeding and the angiography is crucial.