Myxoid soft tissue tumors are an unusually diverse group of neoplasms, most commonly involving the extremities. They are mesenchymal neoplasms characterized by the abundant production of myxoid matrix with a gelatinous appearance. They have varying characteristics in medical imaging and histopathology. The prognosis is variable for recurrence. Less regularly, they are found in bone, skin, the genitourinary tract, aponeurotic tissue, and subcutaneous tissue. We present a case of myxomatous tumor at the scapulothoracic region mimicking bursitis. Myxoid tumors are a heterogenous group of mesenchyme-rich tumors encompassing everything from entirely benign to non-metastasized malignant and metastasized malignant neoplasms. They show considerable overlap in characteristics on clinical examination and on imaging. They are most commonly seen arising from skeletal muscles in the trunk. Their water content is excessive, which gives them low attenuation on CT, high signal on T2-weighted imaging, and low signal on T1-weighted imaging [A 56-year-old male patient had been experiencing pain for six months in his left shoulder. Clinically, two mass lesions were noted on the back side of the shoulder. The largest mass was mobile, with snapping features noted during scapular movement. The MRI showed a cystic lesion in the scapulothoracic region, measuring 8 cm x 1.5 cm. It demonstrated intermediate signal intensity on T1 Figure , and perConsidering the location and signal intensity, the features mimicked scapulo-thoracic bursitis. A US-guided biopsy was done, and the histopathology revealed a myxoid neoplasm. The lesion was planned for surgical excision. No acute or chronic complications were noted. There was no recurrence. Myxoid tumors are a group of malignant and benign lesions distinguished by myriad extracellular myxoid (mucoid) matrixes. They fluctuate in their clinical behavior, ranging from totally safe lesions to locally aggressive and malignant tumors that frequently metastasize. There is a sizeable overlap radiologically and clinically between numerous myxoid lesions, creating a challenge for interdisciplinary teams . The water content of the myxoid matrix is extremely high. Hence, it has a hypoechoic appearance on US, a low signal on T1-weighted images, and a high signal on T2-weighted images, along with a low attenuation on CT imaging. The modality of choice for myxoid tumors is MRI . Differential diagnosis via imaging includes ganglion cysts, nerve sheaths, and other myxoid neoplasms. The well-defined appearance, along with location, perilesional fat or edema, T1/T2 signal intensities, and contrast enhancements, are important in differentiating myxomatous tumors from other lesions ,4. On CTScapulothoracic bursitis, on the other hand, is usually seen as a well-circumscribed cystic lesion between the serratus anterior and the thoracic wall . The sigSince our patient’s mass lesion was in a location that is unusual for myxomatous lesions, the top differential was a cystic lesion, more specifically bursitis. However, a biopsy was done to confirm the diagnosis, which in turn confirmed a myxoid neoplasm. Surgical removal with broad margins is the treatment option of choice with scarce recurrence . Myxomatous tumors are rare in the scapulothoracic region. Therefore, while assessing soft tissue lesions, it should be kept in mind that some lesions that are extremely hyperintense on T2-weighted images can, from time to time, imitate cysts. Imaging, along with a biopsy, is definitively required for the proper diagnosis to differentiate myxomatous tumors from other cystic lesions.


Liver pseudotumors (pseudolesions) pose diagnostic challenges in imaging, often linked to anomalous venous drainage. Notably, aberrant left gastric vein (ALGV) plays a role in segment 2 and 3 pseudolesions. A liver parenchymal pseudolesion due to abnormal venous drainage involving ALGV is highlighted. Understanding hepatic vascular dynamics, exemplified by ALGV-related pseudolesions, aids diagnosis and guides decisions. Further investigating intricate mechanisms underlying these anomalies is crucial.The recognition of third inflow pathways such as ALGV holds significance in distinguishing hepatic pseudolesions from true lesions. Liver pseudotumors, also known as pseudolesions, pose a significant diagnostic challenge in liver imaging. They are characterized by anomalous venous drainage into the liver, including aberrant right gastric venous drainage, parabiliary venous system, epigastric, and cholecystic veins. Among these, aberrant left gastric vein (ALGV) has been identified as a rare entity responsible for pseudolesions observed in liver segments 2 and 3 on post-contrast images .By emphasizing the identification of ALGV-related pseudolesions, our goal is to enhance comprehension of this rare condition, aiding clinicians in precise diagnosis and effective management, supported by diagnostic imaging.A 49-year-old male patient presented with non-specific symptoms, including abdominal pain and bloating. Physical examination revealed no specific findings except mild tenderness upon palpation in the right upper quadrant. Modest derangements in liver function parameters and increased lipid profiles were noted in the patient’s medical records. To elucidate the etiology of the symptoms, an abdominal ultrasound (USG) was conducted. Abdominal USG revealed a hypoechoic solid lesion measuring approximately 5 × 4 cm, situated within segments 2–3 of the hepatic parenchyma. This lesion was observed on a background of fatty liver tissue.Subsequently, a dynamic contrast-enhanced abdominal computed tomography (CT) scan was conducted to provide a more comprehensive evaluation of the lesion. In this dynamic assessment, a hyperdense area was observed in liver segments 2–3, distinct from the surrounding hepatic parenchyma, both in the pre-contrast phase and subsequent phases. While the density of this area was higher than the surrounding parenchyma in the contrast phase, it was similarly contrast-enhanced, resembling the parenchyma. During the venous phase, it was noted that ALGV terminated within this area of the parenchyma. This pseudolesion appearance was attributed to abnormal venous drainage .After this, the patient underwent dynamic magnetic resonance imaging (MRI) of the upper abdomen. The absence of signal loss in the out-of-phase magnetic resonance imaging (MRI) sequence supported the diagnosis of focal fatty sparing. In other sequences of the MRI, a distinct signal was observed within this area due to abnormal venous drainage, setting it apart from the surrounding parenchyma .In this case, we advised lifestyle changes, including a balanced diet, exercise, medical supervision, medication as needed, and avoiding alcohol. Patient education and a collaborative healthcare approach were essential for effective management.Pseudolesions that are challenging to differentiate from liver malignancies or metastases in imaging techniques often arise from two primary factors. These include temporary extrinsic compression, frequently induced by the diaphragm or ribs, and a distinctive third inflow of blood from nontraditional venous sources. ALGV is the cause of this rare third inflow, with a prevalence of 4% .Third inflow, a crucial concept in hepatic circulation, signifies additional blood supply pathways that supplement the primary arterial and portal venous sources. These supplementary pathways, known as third inflow, play a vital role in maintaining adequate liver perfusion. Particularly in scenarios where portal venous flow is compromised, such as portal vein thrombosis, third inflow becomes highly relevant. It ensures continued blood supply to the liver tissue, contributing significantly to overall hepatic function and viability. Recognizing the significance of third inflow provides a comprehensive understanding of hepatic hemodynamics and its implications, especially for conditions like liver pseudotumors associated with anomalous venous drainage .In our case, the discrimination between pseudolesions attributed to ALGV and authentic liver lesions is accomplished by recognizing the distinct anatomical position and evident visualization of these atypical veins traversing regions characterized by heightened contrast. ALGV is classified into three distinct types. Type 1, functions as a pure accessory portal vein, branching out and flowing through the sinusoids. Type 2 vein exhibits a parenchymatous distribution with anastomosis to the portal vein, while Type 3 vein takes a more cranial course with anastomosis to intrahepatic portal vein branches . In our This case report highlights the need to recognize and understand third inflow pathways like ALGV for accurate differentiation between pseudolesions and genuine liver lesions. Awareness of these vascular anomalies aids in precise diagnosis and reduces unnecessary invasive procedures, enhancing patient care. Further research is required to better grasp these complex vascular dynamics and their connection to pseudolesions with anomalous venous drainage.


Sclerosing Mesenteritis (SM) is a rare diagnosis, particularly in pediatric patients, and is typically non-fatal when appropriately treated. Although molecular and immunohistochemical alterations have been described, no pathognomonic signature has been identified for this entity. This report presents a case of a seven-year-old boy who suffered sudden cardiorespiratory arrest. Upon autopsy, he was found to have multicentric SM on the upper mesentery, which led to bowel wall thinning and abdominal bleeding with bacterial translocation. We performed comprehensive morphological, immunohistochemical, and molecular analyses. SM is an atypical disorder with diverse clinical manifestations, including a rare but potentially fatal course. Early diagnosis is critical, given its potential severity. To our knowledge, this is the first case report of pediatric mortality linked to SM. Our findings emphasize the importance of increased awareness and early detection of SM in pediatric patients. To date, no specific incidence of SM reported in pediatric age.3Sclerosing Mesenteritis (SM) is a rare and complex entity with a multifaceted pathogenesis characterized by variable degrees of fibrosis, chronic inflammation, and fat necrosis. The condition was first described by Jura in 1924,4 The first systematic review, conducted by Sharma et al. in 2017,4 included 192 cases and found that SM’s male-to-female ratio was 2.3:1, and a mean age of 55 ± 19.2 years.4 The review also found that symptoms such as intractable abdominal pain, bloating, intestinal obstruction, and massive ascites can last more than a month. Elevated levels of C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR), along with leukocytosis, were the most common laboratory findings.8The literature indicates that SM is more common in Caucasian males with a history of abdominal surgery, trauma, or autoimmune disease.While SM is rarely fatal, this case report presents a unique and unprecedented scenario of a fatal evolution of SM in Pediatrics. The patient's clinical course and symptoms were consistent with previously reported cases, including bowel obstruction and ileus development. However, the patient's condition progressively deteriorated as medical attention was not pursued, leading to a fatal outcome.5 proposed three categories that have become a de facto standard in the absence of gross definitive criteria: Type I, diffuse mesenteric thickening; Type II, single discrete tumor; and Type III, multiple discrete tumors.5 In 1997, Emory et al.6 reviewed 84 cases and proposed histological standards, dividing SM into three categories: mesenteric lipodystrophy, mesenteric panniculitis, and sclerosing (retractile) mesenteritis.6The ambiguity surrounding SM arises from the overlap between variable degrees of fibrosis, chronic inflammation, and fat necrosis. There are two main classifications: clinical pathological and histological. In 1974, Kipfer et al.9The etiopathogenesis of SM remains unclear, but proposed etiologies include infection, ischemia, autoimmunity, paraneoplastic process, and abdominal surgery or trauma. This case report presents an opportunity to deepen our understanding of this exceptional condition’s etiopathogenesis and clinical course, particularly in pediatric age.We present the case of a 7-year-old boy who experienced a sudden cardiorespiratory arrest at home. The patient had a past medical history of ileocolic intussusception at 22 months, which was treated using a sonographic-guided hydrostatic reduction. The family reported several recurrent episodes of abdominal pain over the last few years, but no medical care was sought.On the day of the incident, the patient suddenly complained of abdominal pain and vomited coffee-ground-like material, followed by loss of consciousness. Upon arrival at the emergency, pulseless electrical activity, and severe abdominal distention were depicted. Prompt advanced cardiopulmonary resuscitation (CPR) was started, but nonreactive mydriasis was observed.Further assessment revealed a hemoglobin level of 6.7 g/dL (11.5-15.5 g/dL) and a lactate level of 21 mmol/L, indicating severe anemia and hypoperfusion. An abdominal radiography was performed, which showed pneumoperitoneum. Despite more than 30 minutes of CPR, the patient was pronounced dead.Given the unexpected nature of this event, an autopsy examination was solicited by the Pathology Department.During the corpse’s external examination, multiple bruises of approximately 2 cm in different stages of resolution were observed in the abdomen and thorax, accompanied by lividities. The abdomen was distended and firm by gas or fluid.Upon exposing the abdominal cavity, a malodorous pressure gas was expelled, and approximately 2L of a bloody liquid was found surrounding the intestines. A small bowel loop was observed to be severely damaged without macroscopic perforation .The gross examination of the intestinal loops revealed a retraction and induration of the mesenteric adipose tissue, measuring roughly 5x5cm, beneath the compromised small bowel loop. Furthermore, dozens of lymph nodes and discrete fibrous conglomerates of approximately 1-1.5cm were present in the upper mesentery.Based on the findings, the preliminary cause of death was determined to be the 5x5cm mesenteric adipose tissue retraction area, which caused bowel wall thinning and subsequent abdominal bleeding.The histopathological examination of the mesentery retraction and loop thinning was undertaken in a 20cm section fixated with a buffered-on 10% formalin solution .After fixation, a mid-section cut was performed, revealing a somewhat fibrotic lesion. Representative sections were taken for microscopic evaluation, including the fibrous conglomerates and lymph nodes, which were resected and picked randomly.Following paraffin tissue embedding, 4-micron H&E-stained sections revealed a noteworthy proliferation of fibroblasts over a collagenous stroma, which surrounded and isolated isles of adipocytes, nerves, and vasculature, contacting the muscularis propria with associated extensive areas of hemorrhage. Focal areas of fat necrosis, inflammation, and hemosiderin deposition were also identified. The fibroblasts exhibited fine chromatin without evident nucleoli, pale eosinophilic cytoplasm, and no atypia or atypical mitosis .Similar findings were also observed, but at a smaller scale, along the small bowel mesentery .After a careful macro and micro-evaluation of the lesion, the differential diagnoses were pursued by ancillary studies. The differentials comprised desmoid tumor, familial adenomatous polyposis-associated desmoids, aggressive pediatric fibromatosis, lipofibromatosis, fibrous hamartoma of infancy, low-grade myofibroblastic sarcomas and fibrosarcomas. An immunohistochemistry panel was performed to test for desmin, smooth muscle actin (SMA), caldesmon, beta-catenin, CD-117, DOG-1, S100, Sox-10, CD34, STAT6, IgG4, p53, NTRK, and Ki67 .Among these, only caldesmon, desmin, and SMA tested positive for fibroblast proliferation, and the proliferation index measured by Ki67 was between 1-2%. Molecular analysis was performed by next-generation sequencing to detect mutations in CTNNB1 (exon 3), BRAF codon 600, and hotspots mutation in 40 additional genes, using purified DNA from FFPE-tissue, with no mutations found. Multiplex ligation-dependent probe amplification (MLPA) was used for genes APC, MUTYH, and GREM1, and no copy number variations were detected. The Idylla GeneFusion Assay was used to determine expression imbalance in NTRK1, NTRK2, and NTRK3 genes, which can indicate gene fusion with no expression imbalance detection.Based on the histological examination and the results of the various diagnostic tests, a diagnosis of SM was established.9SM, or retractile mesenteritis, is a rare diagnosis and even rarer in Pediatrics. In our case, the patient was a Caucasian male, which aligns with previous studies, but the underlying cause of SM remains unknown. The primary theories about the etiology of SM center on abdominal trauma, particularly surgical trauma, and an IgG4-mediated autoimmune phenomenon. However, we believe that the ileocolic intussusception the patient experienced does not fit the abdominal trauma category, as hydrostatic reduction is a low-impact procedure that preserves normal anatomy. Full-body X-rays taken during the autopsy ruled out significant skeletal trauma or fractures. Additionally, the autoimmune theory was discarded due to the absence of a familial history of autoimmune diseases, and immunohistochemistry for IgG4 was negative.14Establishing a diagnosis of SM is challenging because it is an exclusion diagnosis. Given the unfortunate circumstances in which we received our patient, the gross examination of the abdominal cavity, and histological appearance, a desmoid tumor was initially considered. However, this diagnosis was ruled out as no small bundles of infiltrative spindle cells in an abundant fibrous stroma were observed. IHC resulted in cytoplasmic for beta-catenin and negative for vimentin, respectively being only positive for SMA in the fibroblasts. As this IHC was not definitive, molecular analysis for CTNNB1 was obtained and found normal. Additional investigation into APC, MUTYH, and GREM1, copy number variations, was conducted, which excluded familial adenomatous polyposis (FAP) associated desmoids and aggressive pediatric fibromatosis.16The lack of fibroblast infiltration beyond the mesentery, the absence of mitotic figures, nuclear atypia, and low Ki-67 directed the case towards a non-malignant entity. Several diagnoses were excluded, including gastrointestinal stromal tumors, solitary fibrous tumors, infantile fibrosarcomas, malignant peripheral nerve sheath tumors, lipofibromatosis, low-grade myofibroblastic sarcoma, fibrous hamartoma of infancy, and desmoid-type fibromatosis. Neither histology nor IHC results correlated with these entities as CD-117, DOG-1, STATE6, CD34, S100, SOX10, and NTRK resulted negative in the fibroblasts, and no fusions were observed in NTRK genes.8 Imaging studies often reveal SM as an incidental diagnosis in CT scans obtained for other reasons, on which a “pseudo-capsule,” a “misty mesentery,” or a more specific “fat ring sign” may be observed. Medical treatments have mainly focused on tamoxifen and prednisone in patients who are not surgical candidates.17 In our case, the patient presented several years before death with moderate abdominal pain as a single symptom, and no CT scans were performed due to his age and mild symptoms. As a result, SM was not diagnosed in vivo, and no surgical or medical treatment was ever considered.SM's clinical presentation includes a wide range of non-specific symptoms, with abdominal pain being the most common and lasting up to 10 years before diagnosis.5 While some studies have noted the association of lymphoma with SM, morphological analysis of the upper mesenteric lymph nodes was histologically normal.9 Our case represents an untreated pediatric Type III retractile SM that naturally evolved into a fatal outcome.5 The observed bowel alterations result from mesenteric fibrosis. This condition progressively obstructs blood flow to the farthest sections of the antimesenteric region. This obstruction compromises the tissue repair mechanisms, thereby creating a vicious cycle that eventually leads to erosion of the superficial layers, rupture of small blood vessels, low-volume hemorrhage, and translocation of bacteria into the peritoneum without any visible perforation.In addition to the primary lesion, other lesions with similar histological characteristics were observed in the upper mesentery. This multicentric presence of SM may align with a Type III Kipfer classification.Our assertions are supported by an array of evidence, encompassing the patient's clinical symptoms, identification of pneumoperitoneum through radiological imaging, and malodorous pressurized gas released during the opening of the abdominal cavity at autopsy. Interestingly, these observations were made despite the absence of any discernible perforations in the gastrointestinal tract.We can only hypothesize about the development of pneumoperitoneum in this case. It is widely accepted that this condition results from a multifactorial process, likely manifesting in the final stages of a multi-year disease. Intense bowel thinning might permit direct gas diffusion into the peritoneal cavity, and the gas diffusion rate could have increased as CPR maneuvers intensified the internal pressure of the small intestines, as illustrated in The most affected areas of the bowel wall exhibited persistent low-volume bleeding, which ultimately accumulated to 2 liters by the time of death. This significant blood loss contributed to a hypovolemic and hypoperfused systemic state. The compromised circulation eventually led to the patient's collapse, characterized by abdominal pain, coffee-ground vomit, and loss of consciousness.Additionally, acute and chronic damage has been documented and is visible in 18Sclerosing mesenteritis is a rare condition with an unclear natural history due to a lack of understanding, inconsistent terminology in the literature, and inadequate follow-up. While cases of spontaneous regression have been reported, the resolution time varies widely. Advances in immunohistochemical and molecular analyses provide an opportunity to gain more insights into the etiology of this condition and its natural history. This case offers a unique opportunity to explore further and understand the natural history of sclerosing mesenteritis, which could lead to better diagnostic and treatment options.20 However, to the best of our knowledge, this report represents the first case of a death that is causally related to SM in a pediatric patient.In recent years, there have been cases of SM in pediatric patients, including an infant who had SM but whose cause of death was not causally related to SM, and a boy in early childhood who underwent satisfactory treatment with Prednisolone.


Spinal arachnoid cysts (SACs) arise either intra- or extradurally and are usually solitary, while cases of multiple SACs have been scarcely reported in the literature. Herein, we report on a rare case of multiple and recurring intradural spinal arachnoid cysts (SACs) causing severe spinal cord compression and neuropathic radicular pain, in a 35-year-old female with a 10-year follow-up. Two separate attempts at surgery were performed but only provided temporary relief since the cysts recurred and new ones formed along the entirety of the spinal cord. Finally, a conservative approach with physiotherapy and a combination of analgesic medications was pursued.The online version contains supplementary material available at 10.1007/s00701-023-05732-1. Spinal arachnoid cysts (SACs) are rare occurrences that have been classified as part of the larger group of meningeal pathologies called spinal meningeal cysts . While sIn this work, we present a case of multiple primary, recurring, and de novo forming intradural SACs in an adult female, with the aim of describing the diagnosis, management, and outcome of this rare occurrence, with a 10-year follow-up. In addition, we reviewed the literature on similar cases and provide a summary of the current knowledge. This report is, to the best of our knowledge, the first to describe the long-term outcomes in a patient with multiple de novo forming and recurring intradural SACs.Written informed consent for both the creation and publishing of the case report with the associated images was provided by the patient in question. The CARE checklist is provided as supplementary (Supplementary file 1).A 35-year-old female presented at her general practitioner’s office with a 3-year history of progressive neck and back pain. She also experienced weakness of the hands and frequently dropped items. Her previous medical history included surgery for a posterior fossa arachnoid cyst at the age of 14. On evaluation, she had radiating pain along the C7 and C8 dermatomes as well as a burning back pain along the level of the scapula, radiating anteriorly. A shooting back pain, just below the inferior angle of the scapula, was provoked by motion. An MRI examination was performed, and the patient was subsequently referred to the university hospital’s neurosurgical department . Under the microscope, the dura was incised and held open with sutures, allowing exposure of the cyst. The arachnoid was dissected sharply, and both cranial and caudal poles of the cyst were identified. The cysts were not adherent to the spinal cord and could be removed in their entirety. Watertight dura closure was achieved using resorbable sutures and fibrin sealant  to support the repaired dura. The laminae were repositioned using microplates . Soft tissues were then sutured in layers to close the wound. The histological analysis confirmed the diagnosis of arachnoid cyst. The patient’s condition improved, and an MRI, 3 months after surgery, showed complete removal of the cysts at this level Fig. . All othTwo years later, the patient developed radicular neuropathic pain of the upper extremities and lower lateral parts of the abdomen. A new MRI was performed in which a marked increase in the number and size of the cysts was found, extending almost continually from C2 to Th11. Spinal cord compression was present along most of this extension. An in-depth review of the current and previous MRIs by the Department of Neuroradiology resulted in the conclusion that there was a marked de novo cyst formation rather than only cyst recurrence. In contrast to the alarming MRI images, the symptoms were relatively mild, and since the cysts had recurred and developed despite two previous surgeries, it was decided to manage the patient conservatively with a trial of analgesic medications. A follow-up MRI was performed 6 months later, showing a stable condition Fig. .Fig. 4SaTwo years later, imaging again revealed a dramatic increase in the size and number of cysts. The cysts extended the entire length of the cervical and thoracic spine Fig. . HoweverSACs, including the intradural and the extradural types, are rare causes of spinal cord compression . TypicalJamjoom et al. reported on a 32-year-old male with five intradural SACs. The patient’s history was also remarkable for lymphedema, distichiasis, Arnold-Chiari malformation, and megaureters, suggestive of the diagnosis of lymphedema-distichiasis syndrome. In fact, cases of frequently recurring intradural SACs were reported in a family diagnosed with this condition . The intOsuka et al. reported on the difficulties in diagnosing a 53-year-old man with multiple thoracic intradural SACs using MRI. Conventional myelography and CT myelography were needed to identify the cyst. Due to the lack of pronounced symptoms, the patient declined the surgery and was instead followed in an outpatient setting .Petridis et al.  reportedZekaj et al. reported the formation of an intramedullary cyst after the removal of multiple thoracic intradural SACs in a 47-year-old female. The authors argue that the longstanding compression of the spinal cord caused by multiple cysts may have led to the formation of an intramedullary arachnoid cyst. In fact, the patient’s intradural SACs were conservatively managed during 3 years before surgical treatment. The authors suggested avoiding delay of treatment of such intradural SACs to prevent worsening prognosis as well as secondary complications such as intramedullary cysts .Hayashi et al. described a 2-year-old with multiple spontaneously disappearing intradural arachnoid cysts . The firIn the case presented, the patient developed several cysts extending intradurally from the posterior fossa to the lower thoracic levels. The etiology of the condition could not be defined. The patient denied any history of CNS trauma, bleeding, or infection and did not report any family history of similar symptoms or conditions. In the absence of these plausible etiologies, genetic factors were hypothesized to be mainly contributing to the condition. However, since the literature lacked reports of any genetic anomaly associated with multiple and recurring arachnoid cysts, the patient did not qualify for further genetic workups, as determined by the Department of Medical Genetics at our institution. Hence, based on these observations, the cysts were considered idiopathic.In the reviewed literature, most patients with multiple intradural SACs were surgically treated. A conservative and expectant management was offered in one case . In our In patients with symptoms of spinal cord and nerve root compression, the diagnosis of spinal arachnoid cysts may be considered. Multiple intradural SACs are rare, with only six cases being reported in the literature, including the one hereby presented. Most patients were offered surgical treatment at the time of diagnosis. However, cyst recurrence may be rapid, and it is important to consider conservative management strategies upon failure of other invasive approaches, especially in the absence of severe symptoms.Supplementary file1 (PDF 1518 KB)Below is the link to the electronic supplementary material.


No complications occurred during and after the procedure. A review of the relevant literature is also provided. Our case is unique, given the therapeutic strategy utilized for pediatric tracheal LCH, and reminds physicians to be aware of tracheal LCH in the differential diagnosis for hemoptysis.Lobular capillary hemangioma (LCH), previously known as pyogenic granuloma, is a benign vascular lesion commonly found within the oral and nasal cavities. However, it is rarely encountered within the trachea, especially in pediatric patients, where it manifests as hemoptysis, cough, and wheeze, and is frequently misdiagnosed as bronchitis or asthma. There is limited literature on the presentation, behavior, and management of tracheal LCH. Herein, we describe a rare case of tracheal LCH in an 11-year-old boy with a history of hemoptysis, which was successfully managed with arterial embolization followed by electrocautery loop snaring  As a beAn 11-year-old otherwise healthy boy presented to our hospital with hemoptysis for 5 days on 20 July 2022. He had more than 10 episodes of hemoptysis per day, with 5–30 ml of bright red blood mixed with clots each time, over the preceding 5 days. Associated fatigue, dizziness, shortness of breath, chest pain, dysphagia, nasal congestion, runny nose, hematuresis, hematochezia, fever, and rigors were denied. There was no history of foreign body aspiration, trauma, hoarseness, airway intubation, or endoscopy. Initially, he was treated at a local hospital for 5 days, but he was not responding well to medical treatment with antibiotics.*4.6*4.6 mm in size affixed to the right wall of the lower trachea  was considered as an alternative option by thoracic surgeons if procedures mentioned above failed, and the extracorporeal membrane oxygenation (ECMO) team was also on standby.On admission, he was in good condition, and the physical examination showed no abnormalities. Thoracic non-contrast-enhanced computed tomography (CT) imaging revealed a cauliflower-like lesion of 4.9 trachea , with an trachea . Flexibl trachea . Glomus In order to minimize the possibility of bleeding during lesion resection, the feeding artery was attempted to be embolized through the right common femoral artery. Digital subtraction angiography (DSA) confirmed the presence of a feeding artery . Under fThen, the tumor was debulked by electrosurgical snaring with a flexible bronchoscope under general anesthesia. In detail, the electrocautery snare was used in a blend mode at 40 W to cut and coagulate the base of the mass , which wIn the present case, the lesion was discovered while searching for causes of non-specific clinical symptoms, such as hemoptysis. It is known that primary tracheal tumors account for <2% of all upper respiratory tract tumors , which aLobular capillary hemangioma (LCH), as benign vascular tumors characterized microscopically by a distinctive lobular arrangement of capillaries , 9, 22, The most common clinical symptoms of tracheal LCH are cough, hemoptysis ranging from minor to massive, and expectoration. Giant masses may narrow the airway, causing dyspnea and breathlessness. The most frequent causes of hemoptysis are infectious diseases, tuberculosis, malignant tumors, cardiovascular disorders, and other inflammatory diseases. Only one of the five kids with tracheal LCH presented without hemoptysis  or a chrThe diagnosis of tracheal LCH relies on chest CT and bronchoscopy, while X-ray may show no abnormality. In detail, chest CT, especially contrast-enhanced CT, identifies the occupying lesions and reveals the nature of the mass with a plentiful blood supply, while bronchoscopy with biopsy plays a key role in achieving the final diagnosis of tracheal LCH and provides additional opportunities for therapeutic intervention. Endoscopic appearances are non-specific and sometimes may be confused with granulation tissue, carcinoid tumor, Kaposi sarcoma, angioendothelioma, paraganglioma, adenoma, angiosarcoma, intravascular angiomatosis, and carcinoma , 8. Hencvia flexible bronchoscopy in an 11-year-old boy was successfully removed in the present case. Collectively, although no adequate guidelines were provided and tracheal LCH is amenable to various techniques, the extent and size of the lesion, as well as patient age and comorbidities require consideration prior to any therapeutic intervention for tracheal LCH.Due to the limited number of tracheal LCH cases, the preferred treatment for it has not yet been established, and therapy methods vary considerably among different studies , 13. Genvia rigid bronchoscopy, and the instillation of cold saline and epinephrine failed to control the massive bleeding was described [20]. Inversely, in another case of BAE followed by lumpectomy under bronchoscopy, the amount of intraoperative blood loss was dramatically decreased [17]. This evidence further emphasizes that BAE was the procedure of choice in order to achieve better control of bleeding.Although our department has rich experience in removing transbronchial tracheal tumors , 24, theMeanwhile, electrocautery loop snaring was used in our case due to its ability to cut and cauterize simultaneously. The specimens obtained by electrosurgical loop snaring for diagnostic purposes are much larger and of excellent tissue quality compared with specimens extracted with forceps. Moreover, alternative surgical options are available. If a flexible bronchoscope fails to remove the tumor, a rigid bronchoscope will be employed. The endotracheal intubation should adjust the depth of endotracheal intubation if the patient loses a substantial amount of blood during the procedure, and balloon compression should be used to try to stop the bleeding. In the event that the endobronchial treatment cannot be performed as intended, the thoracic surgeon will perform a thoracotomy. If vital signs become unstable, the extracorporeal membrane oxygenation team is prepared to provide emergency care. In other words, the ECMO team and thoracic surgeons were on standby as a precautionary measure to ensure the removal of the lesion successfully. This information demonstrates the safe, successful, effective, and less invasive removal of the challenging tracheal LCH without discernible bleeding depends on close interdisciplinary cooperation and elaborate preparation.Since the degree of tumor infiltration into the tracheal wall cannot be evaluated during bronchoscopy, individuals who have undergone bronchoscopy for tumor resection may have residual and incomplete tumor removal, and some patients may experience tumor recurrence. Future monitoring of this patient is required. In other words, it might be necessary to use bronchoscopy during the follow-up period because there is a risk of local recurrence .via flexible bronchoscopy for the diagnosis and treatment of pediatric tracheal LCH with an abundant blood supply. Although exceeding rare, tracheal LCH should be considered a cause of recurrent hemoptysis in children. However, our experience should be validated in further large studies and further rigorous follow-up is needed for the possibility of local recurrence.Our case is unique as it highlights the successful use of DSA/BAE followed by electrosurgical loop snaring The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.Written informed consent was obtained from the minor(s)' legal guardian/next of kin for the publication of any potentially identifiable images or data included in this article.XT and LW collected the data and drafted and edited the manuscript. SL, YWu, CL, EC, ZC, and GJ revised the manuscript. YWa supervised this study. All authors critically reviewed, revised, and approved the final manuscript and agreed to be responsible for all aspects of the study.


Gossypiboma is a rarely reported surgical complication and refers to a retained surgical textile in the body after a procedure. The surrounding inflammation and reaction often manifest as acute pain and subsequently require additional surgery. We report the case of a 33-year-old female who presented with acute abdominal pain one month after undergoing an exploratory laparotomy secondary to a gunshot wound in her home country. A diagnosis of retained foreign body was made with radiological imaging and confirmed upon the retrieval of two surgical sponges after the operation. Due to the high morbidity and mortality as well as increased healthcare costs, strict protocols must be followed to avoid such outcomes. The term “gossypiboma” refers to a retained surgical sponge or textile in the human body. Historically, gossypiboma is a combination of the Latin word “gossypium” meaning cotton and the Swahili word “boma” meaning “place of concealment” . AlthougA 33-year-old female presented to the Emergency Department (ED) of our institution with a one-day history of progressively worsening left lower quadrant and right flank pain. She had suffered a gunshot wound to the abdomen one month ago in a foreign country for which she underwent emergency exploratory laparotomy, right nephrectomy, and ileo-colectomy with primary anastomosis with loop ileostomy. On postoperative day 10, she had chills and diffuse abdominal pain which was determined to be a contained anastomotic leak in the foreign hospital. She was treated conservatively for four weeks with intravenous antibiotics and was discharged with symptomatic improvement. She traveled to the United States once she felt relatively better. Her symptoms had not completely resolved and she presented to our ED with worsening abdominal pain.On a physical examination upon admission, the patient was afebrile, blood pressure was 101/66 mmHg, heart rate was 70 beats/minute, and respiratory rate was 18 breaths/minute. The abdomen was soft with tenderness in the left lower quadrant and right flank without guarding. Right-sided costovertebral angle tenderness was also present. The rest of her physical examination was unremarkable.InvestigationsStaphylococcal epidermidis.Upon arrival, a complete blood count revealed a normal white blood cell count but a decreased hemoglobin value of 10.3 g/dL and a hematocrit value of 32.6%  surgical drains were left in the abdominal cavity. No operative complications were encountered.On day two of admission, the patient was subsequently taken to the operating room for an exploratory laparotomy. Before the exploratory laparotomy, cystoscopy and left ureteral stenting were performed by a separate Urology team. Intraoperatively, a single white lap pad was found in the right nephrectomy bed Figure . Using fPostoperatively, the patient was initially treated with piperacillin-tazobactam and ceftriaxone, but this was adjusted to meropenem 1,000 mg every eight hours after culture sensitivities showed resistance. There were no immediate postoperative complications, and the patient was subsequently discharged on postoperative day five with two JP drains and close follow-up.Gossypibomas have a variable onset and presentation. Gossypibomas have been reported most commonly after abdominal and gynecological surgeries . The timGenerally, the pathophysiology has been described as two distinct inflammatory reactions in the body. If there is an exudative reaction, the patient may present with an acute abscess formation. Alternatively, a granulomatous reaction may cause asymptomatic abdominal adhesions and aseptic fibrous tissue formation ,4. This Imaging modalities such as CT and X-ray do not have consistent radiological features to lead to prompt diagnosis. Gauze that is radiopaque may have a heterogeneous spongiform appearance with air bubbles or calcifications . Over tiIn this case, the patient retained a surgical sponge after her first emergency laparotomy done post-gunshot wound in another country. It is unique in the aspect that there were two separate, recovered surgical sponges in two distinct locations. In many resource-limited foreign countries, there is no availability of radiopaque sponges. This makes imaging to aid in a diagnosis much more challenging. From a public health perspective, gossypiboma is an avoidable potentially fatal surgical complication. Depending on the size of the gauze and duration in the body, the mortality rate has been reported as 11-35% .It is crucial to consider known risk factors, including emergency surgeries, forgoing the preoperative sponge count, staff changes during the procedure, inexperienced staff, and postoperative high body mass index . During Although rare, postoperatively retained foreign bodies in the abdomen remain underreported and represent a serious surgical issue that must be addressed acutely. Its variable presentation can pose a diagnostic dilemma to clinicians. However, identifying risk factors is important and can lead to prompt intervention. CT scan remains the standard imaging modality to aid diagnosis. Some measures that can be taken to reduce the likelihood of this occurring are verifying surgical instrument count, standardizing intraoperative staff hand-off, and encouraging open communication. This report highlights that retained foreign bodies are a continuous underreported medical problem. A gossypiboma must be considered in patients who underwent emergent surgical procedures. Further research must be conducted on optimal operating room communication and environment to prevent such errors.


Introduction: The Buschke–Löwenstein tumor (BLT) is an uncommon sexually transmitted ailment attributed to the human papillomavirus (HPV)—usually the 6 or 11 type (90%)—with male predominance and an overall infection rate of 0.1%. BLT or giant condyloma acuminatum is recognized as a tumor with localized aggressiveness, displaying distinctive features: the potential for destructive growth, benign histology, a rate of 56% malignant transformation, and a high rate of recurrence after surgical excision. There are several treatment choices which have been tried, including laser, cryotherapy, radiotherapy, electrocoagulation, immunotherapy, imiquimode, sincatechins, intralesional injection of 5-fluoruracil (5-FU), isolated perfusion, and local or systemic chemotherapy. In the case of an extensive tumor, preoperative chemotherapy or radiotherapy is used for tumor shrinkage, making the debulking procedure safer. HPV vaccines significantly decrease the incidence of genital warts, also decreasing the risk of BLT; HPV-6 and HPV-11 are included in these vaccines. Materials and methods: We present a 53-year-old heterosexual man, hospitalized in our department in June 2021 with a typical cauliflower-like tumor mass involving the perianal region, which progressively increased in size for almost 7 years. The perianal mass was completely removed, ensuring negative surgical margins. The large perianal skin defect which occurred was reconstructed with fascio-cutaneous V-Y advancement flap. There was no need for protective stoma. The literature review extended from January 1980 and December 2022, utilizing Pubmed and Google Scholar as search platforms. Results: Due to the disease’s proximity to the anal verge and the limited number of reported cases, arriving at a definitive and satisfactory treatment strategy becomes challenging. The optimal approach entails thorough surgical removal of the lesion, ensuring well-defined surgical margins and performing a wide excision to minimize the likelihood of recurrence. In order to repair the large wound defects, various rotation or advancement flaps can be used, resulting in reduced recovery time and a diminished likelihood of anal stricture or other complications. Our objective is to emphasize the significance of surgical excision in addressing BLT through the presentation of a case involving a substantial perianal condyloma acuminatum, managed successfully with complete surgical removal and the utilization of a V-Y advancement flap technique. In the present case, after 5 months post operation, the patient came back with a buttock abscess, which was incised and drained. After another 5 months, the patient returned for difficult defecation, with an anal stenosis being diagnosed. An anal dilatation and sphincterotomy were carried out, with good postoperative results. Conclusions: The surgical management of Buschke–Löwenstein tumors needs a multidisciplinary team with specialized expertise. The reconstruction techniques involved can be challenging and may introduce additional complications. We consider aggressive surgery, which incorporates reconstructive procedures, as the standard treatment for Buschke–Löwenstein tumors. This approach aims to achieve optimal surgical outcomes and prevent any recurrence. The precise incidence and prevalence of BLT are difficult to establish due to a lack of standardized diagnostic criteria; by individually documenting multiple cases, an estimated overall prevalence of 0.1% within the general population is determined, most cases developing between the fourth and sixth decade of life ,3,4,5. AThe literature search encompassed the time span from January 1980 to December 2022 and was conducted on Pubmed and Google Scholar. English was selected as the preferred language for publication. The keywords used were Buschke–Löwenstein and surgery. We excluded articles in non-adult populations. The selection criterion was studies that include clinical data and the therapeutic management  of BLT.Trombetta and Place outlined the clinical manifestations of BLT, with the primary indicator being a perineal mass . DysuriaThe tendency of BLT to infiltrate the surrounding tissues and its high risk for malignant transformation make imaging techniques such as computed tomography and magnetic resonance imaging necessary means for assessing both the local and systemic extent . There are two different perspectives for confirming BLT malignancy: the first consists of neoplastic histological confirmation, and the other is malignant behavior, which indicates the encroachment into neighboring deeper tissues, even in the absence of the histological verification of malignancy. Chu et al. estimate a rate of malignant transformation of 56% . In thei2 laser vaporization, interferon, chemotherapy, radiation therapy, and wide surgical resection, alone or with chemotherapy [There are a lot of treatment options, which include intralesional injection of 5-fluoracil, cryotherapy, interferon, curettage, COotherapy . Despiteotherapy .Lee’s study demonstrated the efficacy and value of a combined regimen involving topical imiquimod, podophyllotoxin, and cryotherapy for managing extragenital giant condyloma acuminata cases, especially when surgical options are constrained by functional considerations ,19.Surgical treatment represented by resection with free margins is the gold standard for BLT. There are many combinations of treatment associated with surgery in an adjuvant or neoadjuvant way, with different results . A unifoThere are multiple surgical approaches for BLT resection, which can be seen as alternatives, such as radiofrequency, electrocoagulation, carbon laser, or classical surgery. Complete resection is very important, with reconstruction at the same time or in a delayed manner . SurgicaIn cases of neoplastic transformation to squamous cell carcinoma in BLT, surgical resection can be coupled with radiotherapy and chemotherapy, either as neoadjuvant or adjuvant treatments. The primary protocols of choice involve the utilization of 5-fluorouracil and mitomycin ,26.2 laser ablation for residual lesions was documented, subsequently paving the way for its integration with other therapeutic approaches [Topical therapies are generally regarded as ineffective due to their elevated failure rate; however, topical imiquimod, which acts as an aminoquinolone modifier of the immune response, has demonstrated advantages in addressing BLT, bucking the trend of ineffectiveness. Initially, a combination of topical imiquimod and COproaches ,24,27.The overall recurrence reported by Chu et al. in their study was 67%; approximately half of the patients who underwent surgical intervention as their primary therapeutic approach experienced a recurrence . The durThe morbidity observed in BLT patients manifests through issues such as the infiltration or infection of soft tissues, recurrences, abscesses, flap failure, fistulas, urethral obstruction, fecal incontinence, anal stenosis, or urinary tract infection .Overall, in the study of Chu et al., the mortality was 21%, associated with morbidities and recurrences, and neoplastic histological transformation did not mean a poor prognosis, having a decreased mortality rate compared to those without the condition [There are no studies which report quality of life associated with BLT. Additionally, the treatment impact, its recurrences, or morbidity are not reported in existing surveys ,29. It iA 53-year-old heterosexual man, a smoker, with no hereditary-collateral background, presented in our department in June 2021 with a typical cauliflower-like tumor mass involving the perianal region, which had progressively increased in size for almost 7 years.  The tumoThe particularity of the case was the presence of a bulky perianal mass (BLT), which was excised with negative surgical margins. The patient developed one complication during the postoperative period, a partial dehiscence of the wound, which was treated conservatively, and two complications in the late follow-up, a buttock abscess and anal stenosis, both of them requiring surgery with a good postoperative evolution.The timely identification of a Buschke–Löwenstein tumor, which primarily hinges on the patient’s consent, along with a dependable pathological diagnosis, is crucial for administering the right treatment and forecasting the treatment results. The surgical treatment of Buschke–Löwenstein tumors requires a multidisciplinary team with special expertise. The reconstruction techniques are difficult and may cause problems such as wound infection, dehiscence, hematoma, or tardive complications, such as anal stenosis or buttock abscess, which may require surgical intervention. There are many non-surgical treatments for this disease, but the majority are not efficient. The gold-standard treatment approach is surgical resection with clear margins and reconstruction in a single operation if the anal canal is involved in less than 50% of the circumference. Additional treatment is unnecessary to prevent postoperative relapses in Buschke–Löwenstein tumors.We regard as standard treatment for BLT an aggressive surgery, which includes reconstructive surgery to uphold the best possible surgical outcomes and avert any recurrence.


Giant arachnoid granulations (GAGs) are poorly investigated. Here, we document clinical findings associated with five new GAGs and illustrate the anatomical composition of these structures as well as diagnostic considerations in three symptomatic adults. The GAGs ranged from 1.1 to 3.6 cm  in maximum dimension and manifested in middle-aged individuals who presented with long-standing brain mass and/or chronic headache. On imaging examinations, the tissues appeared as irregular parasagittal and/or perisinus structures that demonstrated heterogeneous internal elements. The GAGs abutted dura, extended through calvarial marrow spaces, and impinged on dural venous sinuses, causing their stenosis. The histologic workup of two GAG specimens resected from separate individuals revealed central collagen with pronounced internal vascular proliferation. One specimen additionally exhibited reactive changes within the lesion, including venous thrombosis, hemorrhage, and conspicuous inflammation. The salient immune component consisted of a foam cell-rich infiltrate that obstructed subcapsular and internal sinusoidal GAG spaces. Within this specimen, meningothelial hyperplasia was also appreciated. Notably, proliferated lymphatic vascular elements were additionally observed within the structure, extending into deep central collagen regions and engulfing many extravasated erythrocytes in the subcapsular space. In both surgically treated patients, symptoms resolved completely following resection. This report is the first to definitively depict reactive vascular and immunological changes within GAGs that were clinically associated with headache. The frequency of reactive changes within these meningeal structures is unclear in the literature, as GAGs are rarely sampled and investigated. Further systematic analyses are warranted to elucidate the causes and consequences of GAG genesis and their roles in physiology and disease states. Arachnoid granulations (AG), also commonly known as Pacchionian bodies, are present along the meningeal surface of the mammalian brain and are primarily localized in parasagittal brain regions in the vicinity of dural venous sinuses (DVS) . In huma®, Janssen Pharmaceuticals, Inc; Beerse, Belgium) was initiated several years prior to admission but had provided no relief.A patient presented with a complaint of chronic headaches for at least 30 years as well as a history of multiple syncopal episodes. The syncope was sometimes triggered by coughing. The headaches were bitemporal and characterized as a severe pressure sensation that was sometimes pulsating in nature, and occasionally continuous for up to a month in duration. Topiramate  venogram of the head . CT venoEvaluation for intracranial hypertension and potential DVS stenting was recommended.No biopsy specimen was obtained.A patient was admitted for workup after presenting with a complaint of chronic headache. The patient described the headaches as migraines that had been intermittent over several years. Pain episodes began in the occipital region but often radiated toward the forehead. More recently, the patient began experiencing neck pain that radiated into the right shoulder, arm, and first three digits of the right hand. The patient also reported numbness and discomfort in these three digits as well as the radial aspect of the right hand, exacerbated by movement. Additionally, the patient described difficulty with right-sided head movement but denied noticing any masses or tenderness in the suboccipital region. Prior to presentation, the patient had undergone physical therapy, which led to minimal relief of symptoms. The family history was unremarkable, though the patient’s past medical history and review of systems were significant for balance disturbance, sinus headaches, hypertension, and mild memory problems. Physical examination of the head revealed a firm, non-tender, palpable mass in the suboccipital region. A neck exam revealed discomfort and reduced range of motion to the right side. Motor examination revealed minimal weakness in the right brachioradialis and right upper digits, upon digital extension. A sensory exam revealed diminished sensation in the right C6 distribution. Neurological examination was otherwise unremarkable. ® 300) contrast tracer into the GAG interior, which demonstrated prominent internal soft tissue elements.Brain MRI with and without contrast and noncontrast CT of the head  revealedDue to the persistent symptoms and unusual imaging findings, suboccipital craniectomy and surgical excision of the mass were performed and sent for biopsy workup. An intraoperative biopsy of 0.6 to 0.8 cm fragments of fresh tissue returned a diagnosis of benign bone. Titanium mesh cranioplasty was performed. The patient reported significant improvement in the headaches immediately following surgery.A larger resected mass measuring 3.2 × 3.0 × 1.0 cm was received in the pathology lab for permanent section analysis. The specimen was decalcified and then underwent routine processing. On histology, the biopsy material revealed fragments of benign bone and collagen tissue with a proliferation of small blood vessels. The mass was initially interpreted as a cavernous hemangioma, but on subsequent evaluation and radiological–pathological correlation, it was reinterpreted as a GAG. The associated bone fragments exhibited evidence of remodeling and reactive change without evidence of malignancy or other pathological changes. The patient reported complete resolution of the headaches following GAG resection.A patient reported a new onset of unremitting headache and irritation of a chronic scalp “cyst” following an episode of mild closed head injury. The patient had a long-standing history of two presumptively diagnosed GAGs that were present at the skull vertex. At presentation, a large left parietal bone mass was readily palpable on physical examination with intact overlying skin. No meningeal signs or focal neurological deficits were exhibited. Due to the unclear etiology of the symptoms, the patient was admitted for workup of the larger calvarial mass that was associated with the patient’s headaches. Noncontrast head CT revealed cystic diploic expansion of the left parietal calvarium with an associated inner cortical defect, outer cortical endosteal scalloping, and bony remodeling A,B. The At surgery, a circumscribed mass was visualized within thinned calvarial bone following the retraction of scalp skin and periosteum. After outer cortical bone resection, the mass apex was dissected from adjacent bone and an intraoperative biopsy was obtained for smear and frozen section analysis. Hematoxylin-and-eosin (H&E)-stained preparations of the biopsy material revealed bland meningothelial cells arranged in sheets and whorls, admixed with foam cells C,D. IntrRoutine processing and H&E staining of the 2.3 × 1.3 × 0.3 cm resection material revealed fragments of unremarkable bone and periosteum. The associated soft tissue mass consisted of an encapsulated, polypoid tissue A,B. Its Labeling with anti-epithelial membrane antigen (EMA) antibody highlighted meningothelial cells within the capsule and core . LabelinThroughout the lesion, anti-CD1a, pan-keratin, inhibin, kappa, lambda, and amyloid (4G8) antibody labels were all negative, whereas anti-Ki67, CD138, CD45, and progesterone receptor (PR) labels highlighted scant cells only ; S100 highlighted rare nerve twigs in perivascular regions but was negative in the remaining lesion [An extensive panel of special stains including Gram, Grocott’s methenamine silver, Periodic acid–Schiff, and acid-fast bacillus, each performed with deep section repeats on each of two blocks, was additionally negative for microorganisms, and Congo Red stain further confirmed the absence of any amyloid material. GAGs are well documented in radiological literature ,5,6,7 anGiven the prominent and heterogenous nature of the reactive changes identified within this latter specimen , the resection material was further characterized with a comprehensive immunohistochemistry panel. Importantly, labeling with anti-CD31, CD34 and D2-40 antibodies and counterstain revealed that internal vessels within the structure consisted of erythrocyte-containing and erythrocyte-devoid blood and lymphatic vascular channels. D2-40-labeled lymphatic vascular elements within the GAGs consisted of well-formed tubularized lymphatic vessels as well as loosely formed lymphatic channels and singly scattered LECs that haphazardly extended into spaces of the central collagen core .In light of historical literature and generally accepted knowledge regarding human AG structure ,9,10, thAs with asymptomatic non-giant AG, investigation of GAG tissue is challenging in live humans . A recen


Myxoid liposarcoma (MLS), with its risk factors, tends to spread to the lungs and extraperitoneally, with intraperitoneal metastases occurring rarely. We present an unusual case of a myxoid liposarcoma that metastasized to the abdominal organs.A 60-year-old female patient was referred to our hospital for the evaluation of a right upper limb tumor that had been growing for 7 years. The patient refused surgery, and during follow-up, tumor hemorrhage resulted in hemorrhagic shock. The patient’s right upper limb was immediately amputated. MLS was diagnosed histopathologically. Subsequently, the patient underwent adjuvant chemotherapy. Computed tomography (CT) revealed a right buttock mass, a pelvic mass, and left cardiophrenic angle lymph nodes 3 years after the initial surgery. Contrast-enhanced abdominal CT revealed a relatively low-density, lobulated pelvic tumor. Contrast-enhanced pelvic magnetic resonance imaging (MRI) revealed a low-intensity, lobulated mass on T1-weighted images and a high-intensity mass on T2-weighted images. The pelvic mass showed no significant fluorodeoxyglucose (FDG) uptake on positron emission tomography (PET)-CT. On clinical examination, gynecological malignancies were ruled out as the origin of the pelvic lesions. After resection of the right buttock mass, pelvic mass, and left cardiophrenic angle lymph nodes, the patient underwent laparoscopic surgery for a preoperative diagnosis of small intestinal mesenteric metastasis of MLS. A tumor was found in the mesentery of the small intestine and removed with a margin of 5 cm on both the proximal and distal sides. The specimen measured 10 × 8 × 5 cm and contained a multifocal mass. The tumor was found in the mesentery of the small intestine, with no mucosal or submucosal invasion. The patient was diagnosed with MLS with small mesenteric intestinal metastases. On postoperative day 8, the patient was discharged after an uneventful postoperative course. Twelve months after the surgery, there was no evidence of local or distant recurrence.Small intestinal mesenteric metastases of MLSs are rare. Moreover, there are few reports on laparoscopic resection. In this case, the laparoscopic approach was useful in detecting the tumor location and determining the range of resection. Liposarcoma is the most common malignant soft tissue tumor. Myxoid liposarcoma (MLS), a subtype of liposarcoma , generalA 60-year-old female patient presented to our hospital for evaluation of a right upper extremity tumor that had been present for 7 years. The patient declined surgery and developed hemorrhagic shock due to tumor hemorrhage during follow-up. An emergency right upper extremity amputation was performed. Histopathological examination revealed MLS. Subsequently, the patient underwent adjuvant chemotherapy, and two cycles of doxorubicin and ifosfamide were administered for 21 days. Computed tomography (CT) performed 3 years after the initial surgery revealed masses in the left cardiophrenic angle, right buttock, and pelvis. On clinical examination, gynecological malignancies were ruled out as the origin of the pelvic lesions. Abdominal contrast-enhanced CT revealed a relatively low-density lobulated pelvic tumor anterior to the uterus. The small intestine was located adjacent to the mass  showed no significant fluorodeoxyglucose (FDG) accumulation in the pelvic mass. First, the cardiophrenic angle tumor, found in the epipericardial fat tissue with slight adhesion to the pericardium, was resected completely by a thoracic surgeon. Secondly, the right buttock tumor, contained in the gluteus maximus muscle, was resected with 2 cm margins by an orthopedic surgeon. Finally, the preoperative diagnosis was small intestinal mesenteric metastasis of MLS, and the patient underwent metachronous laparoscopic surgery under general anesthesia in the lithotomy position. Initially, diagnostic laparoscopy was performed to evaluate the presence of superficial liver and peritoneal metastases. The tumor was located in the mesentery of the small intestine 60 cm proximal to the Bauhin valve Fig. . The tumThe small intestine was reconstructed using a functional end-to-end anastomosis. The operative time was 118 min, and the intraoperative blood loss was approximately 10 mL. The resected specimen was a 10 × 8 × 5 cm multifocal mass Fig.  with no There are various histological subtypes of soft tissue tumors, with benign tumors accounting for the majority. Liposarcomas are one of the most common histological types of soft tissue sarcoma (STS) . AccordiThere have been reports that more than 95% of MLS has a translocation of the FUS and DDIT3 (CHOP) genes, which is distinct from the well-differentiated and dedifferentiated forms with amplification of chromosome 12q13-15 carrying the MDM2, CDK2, and MHGA2 genes , 9. The The location of MLS differs from that of other STSs. Although most STSs metastasize to the lungs, MLS more commonly metastasize to soft-tissue sites and the spine Table . AdditioLiposarcoma has a higher probability of recurrence than other STSs and differs in the site of recurrence, even in low-grade tumors . The FNCThe effectiveness of whole-spine MRI for follow-up has been described in the National Comprehensive Cancer Network (NCCN) clinical guidelines . FurtherStage I, II, and III disease-free survival rates were 86%, 72%, and 52%, respectively . The oveFurthermore, MLS is recognized as being more radiotherapy-sensitive than other histological subtypes of STS . In addiAlthough recurring liposarcomas require surgical resection, relatively few cases involving laparoscopic resection have been reported . MLS hasSmall intestinal mesenteric metastases from MLS are rare. Moreover, there are few reports on laparoscopic resection. In this case, the laparoscopic approach was useful for detecting the tumor location and determining the range of resection.


A 64-year-old lady with a background of rheumatoid arthritis presented to the foot and ankle clinic with lump underneath the sole of her foot causing significant discomfort. Examination revealed she had a swelling of the first and the second metatarsophalangeal joints. Magnetic resonance imaging revealed abnormal soft tissue thickening between the second and the third metatarsal and a single large encapsulating indeterminate soft tissue mass with a peripheral inflammatory rim. The appearance was suggestive of a malignant sarcoma rather than a rheumatoid nodule or rheumatoid tenosynovitis. The patient was referred to the regional sarcoma unit where the scans were reviewed, and a sarcoma was ruled out. The patient then underwent excision of the indeterminate soft tissue mass. Histology revealed granulomatous infiltration suggestive of a rheumatoid nodule. This has not been described previously in the literature. Rheumatoid nodules are well-demarcated, flesh coloured, subcutaneous lumps occurring in a quarter of patients with rheumatoid arthritis . The mosA 64-year-old lady with a background of rheumatoid arthritis presented to the foot and ankle clinic with lump underneath the sole of her foot causing significant discomfort. She had a background of anticitrullinated protein antibody positive rheumatoid arthritis treated by oral methotrexate (MTX). In the past she had a surgical fusion of the right foot first metatarsophalangeal joints (MTPJ) as a Weil’s osteotomy of the second toe metatarsal. On examination she had painful soft tissue mass swelling over the first and the second MTPJ cystic in consistency.Magnetic resonance imaging revealed abnormal soft tissue thickening between the second and third metatarsal and a single large encapsulating indeterminate soft tissue mass with a peripheral inflammatory rim. The appearance was suggestive of a malignant sarcoma rather than a rheumatoid nodule or rheumatoid tenosynovitis. Laboratory test showed systemic inflammation with C-reactive protein levels of 64 mg/l (reference range <5 mg/l), normal serum urate  and blood cultures were negative.The patient was referred to the regional sarcoma unit where the scans were reviewed, and a sarcoma was ruled out. The patient underwent surgical excision of the indeterminate soft tissue mass. Intraoperatively, a large soft tissue mass 4.0 × 3.0 cm in diameter involving the digital nerve and capsule of the second MTPJ was noted, along with necrosis of the underlying fat pad and 2.Histopathology concluded the specimen consisted granulomatous infiltration with peripheral palisading and no evidence of features specific to a neuroma, dysplasia or malignancy. The presence of granulomatous lesions was indicative of a rheumatoid nodule with local infection.Post-operatively at the 3 month follow-up the patient was able to ambulate pain free with no distal neurovascular compromise. She was subsequently discharged from the clinic.Rheumatoid nodules occur in ~20% of all adult rheumatoid arthritis patients . Most paOn plain radiograph, the subcutaneous rheumatoid nodule appears as a lobular expansive soft tissue mass. Lesions rarely contain calcifications, and it can be a differentiating point from a mass. In addition, it is known that cases of bone erosion caused by rheumatoid nodule are rare . Recentlet al. [El-Noueam et al.  classifiet al. [Starok et al.  reportedet al. .Magnetic resonance imaging findings of rheumatoid nodules are not specific, but considering the patient’s history and clinical features, the width can be narrowed, and it can be used to evaluate the range before surgery during surgery. Indications for rheumatoid nodule surgery include pain, progressive neurological abnormalities, decreased joint mobility, cosmetic problems and differentiation from soft tissue masses.In this case, there was a history of rheumatoid arthritis, but the symptoms were not severe, and it was clear that the rheumatoid factor was positive, and an abnormal soft tissue thickening between the second and the third metatarsal and a single large encapsulating indeterminate soft tissue mass with a peripheral inflammatory rim was detected on magnetic resonance imaging.et al. [It was a mass with suggestive findings and other nonspecific findings, making it difficult to differentiate it from synovial sarcoma or soft tissue sarcoma such as malignant fibrous histiocytoma. The possibility of granulomatous synovitis caused by tuberculosis may be considered a differential and proliferative pigmented villous nodular synovitis. In addition, even if the symptoms are not severe, it is considered that rheumatoid nodule should have been included in the differential diagnosis considering the medical history. Isdale et al.  presenteIn patients reporting pain and presenting with an indeterminate soft tissue mass located in the sole of the foot, we urge clinicians to consider the referral to the regional sarcoma unit to rule out possibility of sarcoma as the primary diagnosis.Although not specific, rheumatoid nodules can be used for the differential diagnosis of solid or well-circumscribed subcutaneous tissue masses with internal necrosis that occur in areas where there is a lot of friction between bone and skin in patients with rheumatoid arthritis.This case is rare, at the time of writing there are only a handful of published papers detailing a rheumatoid nodule presenting as an indeterminate soft tissue. Our hope is that this information can be used by surgeons to inform their decisions when referring to the regional sarcoma unit and improve patient outcomes.


Spinal meningiomas (SMs) are a prevalent subtype of central nervous system tumors, with the majority adhering to the dura mater. In this case, we present the case of a 72-year-old female who initially reported numbness in her legs and the gradual onset of gait disturbances. Over a three-week period, these symptoms progressively worsened until she experienced a sudden onset of weakness and neurological deficits, leading to the diagnosis of acute cord syndrome (ACS). Magnetic resonance imaging revealed an anomaly within the extramedullary space, precisely located at the T8-T9 level. This anomaly exhibited peripheral gadolinium enhancement and demonstrated a dural tail sign, indicating the presence of an abnormal mass. Furthermore, a dorsal spine CT scan confirmed these findings by revealing a hyperdense lesion localized within the T8-T9 region. The lesion was situated posterior to the spinal cord, and conspicuous alterations in the coloration of the dura mater at the corresponding level were evident. A complete surgical resection was performed successfully, and the patient's surgical intervention proceeded without complications. Following the surgery, we observed significant improvements in both sensory and motor functions compared to the patient's preoperative state. Acute cord syndrome (ACS) presents a neurological emergency marked by the abrupt onset of spinal cord dysfunction, often arising from spinal cord compression or ischemia . One lesThe usual way SMs manifest clinically is with pain as the initial symptom, which is subsequently followed by issues related to walking, sensory function, and bowel or bladder control . HoweverThis case report examines ACS caused by SM, offering insights into its clinical manifestations, diagnostic challenges, management complexities, and implications for patient outcomes. By shedding light on this uncommon yet potentially devastating condition, this case report aims to contribute to the growing body of knowledge surrounding ACS due to SM and underscores the importance of timely diagnosis and intervention in achieving favorable patient outcomes.A 72-year-old female was admitted complaining of persistent pain and numbness in her legs for three weeks and a recently developed gait disturbance. Additionally, the patient reported experiencing severe nocturnal back pain that appeared to have a crescendo pattern. However, the most striking observation was the acute onset of significantly exacerbated symptoms in the past 24 hours, rendering her entirely dependent on assistance for ambulation. There were no significant prior medical conditions or injuries in her history. A neurological assessment disclosed bilateral numbness in the lower limbs that was particularly left-side dominant. The patient exhibited a distinct sensory level on the right side, corresponding to the T10 dermatome. On the left side, the sensory level extended more cephalad, encompassing the dermatomal distribution up to T5. These findings suggest an asymmetrical sensory deficit, with a clear demarcation at the midline, and a higher level of sensory involvement on the left side compared to the right. Hip flexion and extension were motor grade 1/5 and 0/5 on the right side and left side, respectively. Knee flexion and extension were also motor grade 1/5 and 0/5 on the right side and left side, respectively. Ankle dorsiflexion and plantar flexion were motor grade 2/5 on the left side and 4/5 on the right side. Sensations were severely affected bilaterally, but more so on the left side. Bilateral Babinski and ankle clonus reflexes were positive. Bladder and anal sphincter functions remained intact, and there were no issues with the motor and sensory functions in the upper extremities. Plain radiography of the cervical and thoracic spine did not reveal any notable abnormalities. Magnetic resonance imaging (MRI) demonstrated a lobulated mass in the T8-T9 dorsal space, particularly in the intradural extramedullary space. T2-weighted MRI showed a low-signal-intensity elliptical lesion, which was enhanced peripherally on a gadolinium-enhanced T1-weighted image (Figure The T7-T9 spinous processes were removed. A bilateral T8-T9 thoracic laminectomy and a vertical incision in the dura were performed. The arachnoid was dissected, and the lesion was visualized posterior to the cord. A microsurgical approach was employed to carry out meticulous resection and debulking (see Figure The histopathological examination of the tissue specimen, consisting of a single piece of grayish tissue measuring approximately 1 × 1 × 0.5 cm, definitively identified the presence of features in alignment with those indicative of a grade 1 meningioma, as depicted in Figure The leg numbness and gait disturbance displayed gradual improvement in the postoperative period. Upon assessing the patient postoperatively, we observed a remarkable enhancement in both lower limb motor and sensory functions compared to their preoperative state. Specifically, the motor strength in the right lower limb had progressed to grade 4/5, while the left lower limb demonstrated an equivalent motor grade of 4/5. This substantial improvement in motor function signifies a significant recovery in muscle strength in both lower extremities. Moreover, sensory evaluation revealed noteworthy improvements, indicating a substantial recovery of sensations that had been compromised before the surgery. This overall enhancement in motor and sensory functions postoperatively underscores the success of the intervention and the potential for an improved quality of life and mobility during the rehabilitation process.One week following the surgical intervention, the patient's postoperative recovery has shown promising progress. Of particular note is the substantial improvement in the patient's gait disturbance, which has significantly ameliorated since the procedure. Encouragingly, the patient now exhibits the ability to move independently without requiring external assistance or support. This remarkable achievement underscores the favorable trajectory of the patient's postoperative rehabilitation, reflecting the effectiveness of the surgical intervention and providing optimism for continued recovery and improved mobility in the days ahead. A postoperative MRI was deemed unnecessary, as the surgical procedure successfully achieved complete resection of the lesion, with no indications or suspicions of any residual tumor remaining.ACS is characterized by the sudden onset of neurological deficits resulting from spinal cord compression or ischemia . In the The management of ACS caused by SM requires a multidisciplinary approach involving neurosurgeons, neurologists, and rehabilitation specialists. Prompt surgical intervention is the first-line therapy for SM and is generally considered a safe and efficient procedure in experienced centers. The rate of complete resection of SM mentioned in the literature ranges between 82% and 98% . This inThe presented case demonstrates that ACS due to SM, though rare, demands swift recognition and intervention. When diagnosed early in its course and managed appropriately, patients have the potential to experience substantial improvements in neurological function, highlighting the critical role of multidisciplinary care and surgical expertise in optimizing patient outcomes. Further research and the sharing of clinical experiences will continue to refine our understanding and management of this complex condition, ultimately benefiting patients facing this challenging diagnosis.


Pleomorphic liposarcoma is the rarest subtype of liposarcoma. Pleomorphic liposarcomas are generally unresponsive to chemotherapy and radiotherapy. Moreover, metastasis in the liver, as the first and sole site, from a primary extremity soft tissue sarcoma, including pleomorphic liposarcoma, is extremely rare. Information regarding the appropriate management of these lesions is limited.A 50-year-old Japanese woman presented with a mass in the left thigh. Imaging examination revealed a soft tissue sarcoma on the left posterior thigh. The tumor was histologically diagnosed as pleomorphic liposarcoma. Computed tomography examination for assessment of metastases incidentally detected a huge liver mass. Wide excision of sarcoma was performed prior to chemotherapy. Right trisectionectomy was necessary to achieve hepatic clearance; however, the future liver remnant volume was insufficient. Therefore, we decided to administer anthracycline-based chemotheraphy to shrink the tumor. After seven courses of adriamycin-based chemotherapy, the liver tumor size was reduced from 211 mm × 106 mm × 180 mm to 105 mm × 66 mm × 90 mm. Finally, a right hemihepatectomy was performed. The patient was continuously monitored and was metastasis or local recurrence free within 5 months after liver surgery.Chemotherapy is effective in some cases for the treatment of unresectable liver metastases of pleomorphic liposarcoma, and complete resection is possible with conversion surgery. If the patient’s general condition permits, anthracycline-based chemotherapy can be used for the treatment of stage 4 pleomorphic liposarcoma. The World Health Organization defines pleomorphic liposarcoma (PLPS) as a pleomorphic, high-grade sarcoma containing a variable number of pleomorphic lipoblasts. PLPS is the rarest subtype of liposarcoma, accounting for approximately 5% of all liposarcomas . It commWe report the case of a 50-year-old Japanese woman who visited a local hospital owing to a palpable mass on the left thigh. The mass gradually increased in size and was associated with left thigh pain. The patient was referred to our hospital after 8 months. The patient had a history of hepatitis C virus (HCV) carrier, but no other major preexisting medical conditions were noted. Magnetic resonance imaging revealed the presence of sarcoma on the posterior left thigh Fig. a, b, andAt 4 weeks postoperatively, the metastatic liver tumor had increased to 211 × 106 × 180 mm in size  had metastasis to sites other than the lungs as a first or single occurrence. Of them, one patient developed liver metastasis from a chondrosarcoma, while liver metastasis did not occur in the only patient who had soft tissue sarcoma [The lungs are the most common metastatic site. Hepatic metastases from primary soft tissue sarcomas frequently present with visceral and retroperitoneal involvement . Liver m sarcoma . The his sarcoma . However sarcoma , while t sarcoma . To dateet al. reported OS times of > 3 years in patients who were not potential candidates for metastasectomy, but less than 6 months in those who were inoperable and treated with chemotherapy [By contrast, metastasectomy for removal of metastatic soft tissue sarcoma in the liver prolongs the prognosis . The preotherapy . Chemothotherapy . Some stotherapy . In thisIn conclusion, we report herein an extremely rare case in which a patient with PLPS in extremity developed liver metastasis without lung metastasis at the first visit. Moreover, we report the first case of PLPS in extremity with hepatic metastasis treated with adjuvant chemotherapy and conversion surgery of liver resection. This case demonstrates the potential efficacy of chemotherapy and conversion surgery in treating unresectable metastatic liver tumors with primary soft tissue sarcoma.


Tanycytic ependymoma has been marked as Grade II by the World Health Organization (WHO), requiring considerable treatment. However, according to the fifth edition of the WHO Classification of Tumors of the Central Nervous System published in 2021, tanycytic ependymoma is no longer identified as a subtype of ependymoma. Herein, we offer an unusual case of a supratentorial ependymoma, previously tanycytic ependymoma. Which radiologically mimic pineal region tumors; however, they pathologically mimic meningiomas, schwannomas, medulloblastomas, or astroblastomas.A three-year-old girl presented to our neurosurgery department with sudden onset gait disturbance and balance impairment; we detected no additional neurologic deficit. Magnetic Resonance Imaging (MRI) revealed a giant, multilobulated, well-circumscribed right pineal mass, approximately 4.5 x 4.5 x 4.5 cm in size, crossing the midline and extending posteriorly, invading the pineal region. The initial diagnosis was a pineal region tumor. Following gross-total resection of the tumor, pathology reports showed tanycytic ependymoma. Postoperatively the patient's gait disturbance was improved, and there was no balance impairment. Follow-ups at three and six months, no sign of recurrence has been encountered.Our case demonstrates that supratentorial ependymomas may also occur in the pineal region and requires an accurate neuropathologic diagnosis. Early accurate diagnosis is essential; since those tumors may be related to a wide range of prognoses and necessitate different treatment modalities. Ependymomas are one of the most common malignant intracranial neoplasms in children. Although they can arise in practically every area of the neuroaxis, the posterior fossa is the most frequent location, followed by the supratentorial region and the spinal cord . We discA three-year-old girl with no history of congenital defects or comorbidities presented to neurosurgery with gait abnormalities and rapid balance impairment, which had developed over four months. Following a physical examination, we detected no further neurological impairment. Non-contrast-enhanced computerized tomography (CT) showed a midline isodense intracranial lesion, including hyperdensities near its center, approximately 4.5 × 4.5 × 4.5 cm in diameter, in the pineal region next to the quadrigeminal cistern Figure Subsequent contrast-enhanced magnetic resonance imaging (MRI) revealed a calcified hemorrhagic, well-circumscribed lesion in the right pineal region and isointense on T1-weighted sequences Figure . Both laAfter a right parieto-occipital craniotomy, the tumor was grossly totally resected. Macroscopically, the tumor was a well-circumscribed, slightly firm configuration, a gray-colored mass that crossed the midline and extended posteriorly, invading the pineal region. Although it was highly adherent to the surrounding vascular structures and parenchymal tissues, gross-total resection was accomplished Figure . The vasPathologic examination revealed a 44 cc volume of cream-yellow curettage material from the mass spreading from the midline to the pineal region at the third ventricle level. Light microscopy showed that the tumor consists of spindle cells with oval and elongated nuclei with speckled chromatin and small nucleoli arranged in interlacing fascicles  and showed characteristic perinuclear dot-like positivity within the cytoplasm with epithelial membrane antigen (EMA)  was gross-totally resected, and the patient received no postoperative radiochemotherapy .On the other hand, a study group from India presented a rare case of anaplastic ependymoma  of the pineal region in a 42-year-old female patient. First, they performed ventriculoperitoneal shunt placement, then excised the tumor. They achieved a gross-total resection, and the patient underwent postoperative radiotherapy . FurtherAn unusual case of substantial calcified supratentorial ependymoma was presented in the pineal region of a three-year-old child. The ependymoma radiologically mimicked other pineal region tumors. Gross-total resection should be the treatment of choice. This case underlines the significance of early and accurate diagnosis and surgery to achieve an effective outcome and plan appropriate follow-up treatment. Further studies are required to hypothesize their radiological resemblance to midline tumors. When a sizeable centrally calcified midline lesion is observed on imaging, a surgeon should carefully evaluate the case, and ependymomas should also be considered in the differential diagnosis.


Prolactinomas are benign pituitary tumors also known as prolactin-secreting adenomas (PSA). These tumors cause excessive secretion of prolactin (hyperprolactinemia), a hormone responsible for lactation. Diagnosing hyperprolactinemia relies on measuring prolactin levels in the blood, and elevated serum levels of prolactin are typically indicative of prolactinoma. The hook effect occurs in immunological tests such as the prolactin level test. When the amount of prolactin present in the sample is too high and exceeds the binding capacity of the antibodies being used, the test result may indicate falsely low levels of prolactin, which is the hook effect. The present study describes the case of a male patient who presented with neck pain and difficulty swallowing. MRI revealed a giant (>40mm) extradural tumor affecting the clivus, anterior fossa, pterygopalatine, and bilateral infratemporal fossae as well as the petrous apex and bilateral cavernous sinuses. Endocrinological investigation yielded no specific abnormalities. An occipitocervical fixation (arthrodesis) was proposed with simultaneous extended endoscopic endonasal resection. Surgery succeeded in resecting a portion of the clival tumor and the anterior fossa. Measurement of prolactin levels several weeks post-surgery found them to be extremely high, confirming the hook effect. Prolactinomas are benign pituitary tumors also known as prolactin-secreting adenomas (PSA). These tumors are more common in women of childbearing age and have an incidence of three to five per 100,000 inhabitants; however, these values may vary depending on the region . HoweverThe hook effect is seen in laboratory assays using antibodies to detect and quantify a substance ,5 and whTo mitigate the hook effect and obtain accurate prolactin measurements, dilution of the blood sample is recommended. Dilution reduces the concentration of prolactin, thus enabling accurate measurement. In suspected cases of the hook effect, healthcare providers may request a dilution study or employ alternate testing methods, such as serial dilutions or other specialized assays, to confirm the diagnosis of hyperprolactinemia -6.A male patient presented with a giant extradural tumor affecting the clivus, anterior fossa, pterygopalatine, and bilateral infratemporal fossae, as well as the petrous apex and bilateral cavernous sinuses Figure . The patThe patient underwent occipitocervical fixation (arthrodesis) Figure . SimultaThe surgery was successful, and the patient was discharged without a nasoenteral tube after evaluation by a videofluoroscopic swallowing study and laryngoscopy, which demonstrated effective swallowing without bronchoaspiration. An MRI performed 24 hours after surgery confirmed total resection of the clival component and the anterior fossa involving the internal carotid arteries, as planned prior to surgery Figure .A histopathological examination and an immunohistochemical study, together with the MRI findings, confirmed the diagnosis of invasive adenoma arising from prolactin-producing cells, or macroprolactinoma. Prolactin was positive in 100% of the cells and Ki-67 was positive in 10% of the cells Figure .Subsequent measurement of prolactin levels weeks after the surgery was found to be at 470 Ng/ml, thus confirming the hook effect.Based on this diagnosis, cabergoline was started and a positron emission tomography (PET) scan was done. There was no tumor present elsewhere, confirming the diagnosis of invasive adenoma rather than carcinoma. After the pharmacological treatment, prolactin levels dropped to 170 Ng/ml.The hook effect is observed in immunological tests, such as when measuring serum levels of prolactin, where the results can be falsely low due to excessive antibody saturation . This ocInitially, prolactinomas are pharmacologically treated using dopaminergic agonists. The first-choice drug is cabergoline with an initial dose of 0.25 mg twice weekly as a starting dose, increasing by 0.25 mg per week, up to 1 mg orally twice weekly in four weeks, maintaining as the serum prolactin level drops .Previous studies have addressed the hook effect in different clinical contexts ,10. For Recent studies have investigated alternative approaches to overcome the hook effect and obtain more accurate results ,12. For A pituitary adenoma is very hard to diagnose as it can mimic many other conditions which are often the first choice of diagnosis during initial examinations. Confirmation can also be a challenge, so differential diagnosis of prolactinomas in particular must be considered, as follows: (i) Esthesioneuroblastoma (ENB), which are rare, malignant neoplasia that originate in the olfactory neuroepithelium. They typically grow toward the upper nasal cavity and may extend into the paranasal sinuses or intracranial region. ENBs are characterized by their neuroectodermal origin and can display a wide range of histological patterns ; (ii) MeIn 2022, the 2021 World Health Organization Classification of Central Nervous System Tumors  and 2022This case exemplifies a patient who initially presented with a prolactin level lower than 20 Ng/ml and, after surgical resection, began to exhibit values above 450 Ng/ml. Given the difficulty encountered in the initial evaluation, it is crucial that in similar cases, endocrinological evidence is also sought.The hook effect is a significant phenomenon to consider in the diagnosis and monitoring of giant and invasive prolactinomas. Understanding this effect and adopting alternative testing methods are essential to avoiding misdiagnoses and ensuring proper evaluation of hormonal status in these cases.