Carcinoid tumors have been treated with formal oncological surgical resection, which is considered the gold standard approach. In the past two decades, bronchoscopic has gained popularity for treatment of carcinoid tumors. A 34-year-old female with an unresolving chronic cough, underwent a chest CT that showed right endobronchial lesion. Bronchoscopy and a biopsy were taken from the lesion; pathology confirmed the diagnosis to be a typical carcinoid tumor. Forty-six-year-old male, who was a smoker, suffered from hemoptysis. Pathology revealed it to be an endotracheal typical carcinoid tumor at the carina. Uncertainties, such as chances of recurrence, cost-effectiveness and the inability to achieve formal oncological resection have been discussed and challenged in the literature. With gaining use of this treatment approach, tissue-sparing surgery for carcinoids can improve patient quality of life both in the short and long term. Both tend to have an endobronchial location, either centrally or peripherally [et al. presented an algorithm for management of carcinoid that starts initially with bronchoscopic treatment, advocating a tissue sparing approach, and arguing against extensive surgical resection early on, pointing towards the lack of evidence to support the notion of advancement of the disease to the point where a more radical resection is warranted, given the slow-growing nature carcinoids in general. The authors reported a 42% rate of complete remission in a sample of 112 patients with carcinoid cancer; with a disease-specific 5-year survival rate of 100% following their approach [Tracheal and bronchial typical and atypical carcinoid tumors, belonging to the subset of thoracic neuroendocrine tumors that also is comprised of small cell lung carcinoma and large cell neuroendocrine carcinoma, have had a paradigm shift in its treatment approach; moving towards tissue-sparing endobronchial resections with close monitoring from the gold standard of formal surgical oncological resections . Referripherally . Brokx eA 34-year-old female, who is a known case of ulcerative colitis, has been followed by a pulmonologist due to a chronic cough, that was first treated as asthma; but did not resolve with the management plan set by the pulmonology service. A CT scan  was thenThe second case involved a 46-year-old male, who was a smoker for 15 years, however, had quit smoking for 1 year prior to presentation, otherwise medically and surgically free. He had a main complaint of periodic hemoptysis. This patient’s CT scan  showed aBoth patients were followed 3 months later with a repeat bronchoscopy, which was clear from any signs of recurrence.See The use of a tissue-sparing resection strategy for endotracheal and endobronchial carcinoid tumors has been challenged by the notion of inability to achieve an oncological; that is by surgical lymph node resections and by resecting beyond the tumor’s gross borders, satisfying the possibility of an extraluminal tumor growth. For the past two decades, questions have been raised regarding initial bronchoscopic resection of carcinoid tumors, such as whether a less aggressive approach to treatment would lead to a worse outcome later on if recurrence occurs, and whether this strategy would be less cost-effective without a definite clear advantage to the gold standard oncological surgical resection. Surveillance and follow up algorithms also play a crucial role in patients undergoing initial bronchoscopic resection, and a consensus has yet to be set.et al. reported a success rate of 74% in a group of nineteen patient who underwent bronchoscopic resection, with the remainder undergoing radical surgical resection upon follow up. The authors suggest following patients utilizing a high-resolution CT scan to assess for extraluminal extension after initial bronchoscopic resection in patients who are fit for minimally invasive debulking surgery [Van Boxem  surgery .et al. published findings of a prospective long-term study after initial bronchoscopic resections of carcinoid tumor that showed a recurrence rate of 7.8% in a group of 51 patients who were diagnosed 47–198 months after initial resection, noting the distant metastasis in some. However, the author argued that the decision of initial bronchoscopic treatment did not compromise the later need for radical surgical resection [Brokx Typical carcinoids have a low tendency to metastasize to lymph nodes, unlike atypical carcinoids; 3–20% versus 48–75%, respectively . Since tet al. concurs on the initial tissue-sparing strategy; however, raising the point that this might not be suitable in developing countries, where patients usually present late with a more complicated disease progression in comparison with the experiences of surgeons in more developed countries. Feasibility of follow-up can also be questioned in the absence of a technologically advanced health care system [Dewan e system .In Saudi Arabia, surgical strategies vary when dealing with carcinoid tumors. While metropolitan cities can follow a tissue-sparing strategy with ease considering the availability of specialized thoracic surgeons, technologically advanced tertiary care hospital networks, and a health-aware patient population, the same cannot be generalized to more rural areas where access to niche surgical care may be limited. Taking this into consideration, it is integral to inquire about patients’ access to healthcare and general background before opting for initial bronchoscopic resection with close follow-up. Moreover, follow-up algorithms have yet to be well described in the aforementioned strategy, which also presents itself as a challenge in this developing movement of tissue-sparing surgery. Mode of resection being by endoscopic electrocautery, snare resection, the use of a laser modality have not been compared in terms of recurrence, whether any would have an effect on recurrence and achieving adequate excision. It is exciting to consider tissue-sparing resection being the mainstay of treatment considering the benefits it offers the patient when it comes to limiting post-operative complications that accompany radical surgical resections, improving quality of life and hastening return to daily activities. Prospective studies need to be performed in a multicentric fashion to be able to fully answer the questions around this bold approach to treatment of carcinoid tumors.Carcinoid tumors that are endotracheal and endobronchial managed with bronchoscopic resection showed promising post-operative and short to medium term results in decreasing patient morbidity and allowing for quicker recovery; with gaining popularity of this approach, there is hope in a robust long term follow-up model to streamline tissue-sparing surgery for carcinoids of the trachea and bronchus.S.A. had the main role of literature review and analysis of relevant papers, writing the manuscript and supervision of all related roles in producing this study. H.A. initiated the study, revised and reviewed the manuscript. M.A. had an advisory role and is the director of this initiative of tissue-sparing surgery in Saudi Arabia. R.A. aided in writing the manuscript, gathering data and final review.The authors declare that there are no competing interests in regards to this paper.


Low-grade fibromyxoid sarcoma is an extremely rare malignant neoplasm, with an incidence of 0.18 per million, and comprises 0.6% of all soft tissue sarcomas. It has a high recurrence rate and late metastatic spread and is chemotherapy and radiotherapy insensitive. This paper reports a case of an unusually large mediastinal low-grade fibromyxoid sarcoma in a 55-year-old patient. The tumor was engulfing the main blood vessels of the mediastinum, involving the lung, and extending beyond the chest cavity to involve the cervical and axillary regions. The patient has a 21-year history of frequent surgical resections for lesions that were repeatedly misdiagnosed as neurofibroma. The tumor was successfully resected by a challenging operation that involved mediastinal mass resection, chest wall mass resection, and wedge resection of the left upper lobe of the lung. The deceivingly benign-looking histology of this tumor makes it a commonly misdiagnosed one, requiring careful assessment by pathologists to reach the right diagnosis. Surgical resection with clear margins remains the treatment of choice for these lesions. Due to the behavior of this tumor, once detected and managed, extensive long-term follow-up is always recommended. Low-grade fibromyxoid sarcoma (LGFMS) is a rare soft tissue malignant tumor with relatively benign histological characteristics, late metastatic potential, and an alternating fibrous and myxoid background . Evans oMost LGFMS tumors are deep-seated lesions originating from soft tissues in the proximal limbs and torso; they usually grow slowly and present as a nontender mass. Grossly, the tumor has a well-defined white appearance. Microscopically, the cells have a spindle to stellate appearance, with small, angulated nuclei and little cytoplasm . CommonlGenetically, chromosomal translocations leading to fusion of the FUS and CREB3L2 genes are associated with most cases of LGFMS. FUS-CREB3L1 fusion is also reported with a lower incidence. However, some tumors present with neither translocations .LGFMSs are distinguished by their high potential for metastatic spread and recurrence, with the lung being the most common site of metastasis. Owing to their low mitotic activity, LGFMSs are not considered sensitive to chemotherapy or radiotherapy and, therefore, are not used in treatment. Surgical excision remains the mainstay of treatment , 3.Here, we present a case of a huge low-grade fibromyxoid sarcoma engorging the main mediastinal blood vessels with neck and axillary involvement and infiltration to the left lung, necessitating wedge resection of the upper lobe. The patient was complaining of dyspnea on mild exertion, orthopnea, and dysphagia to solids. This case was repeatedly misdiagnosed for over 21 years as a neurofibroma and was correctly identified only after her most recent resection surgery at our center. The patient’s treatment course included multiple resections over the mentioned period.A 55-year-old female patient was admitted to our hospital, complaining of shortness of breath on mild exertion of 5 months.via median sternotomy. The acquired pathology report from that time indicated a neurofibroma. Three months later, she noticed a lateral neck mass of about 5 × 5 cm, which was resected.The patient’s history goes back 21 years ago when she noticed an anterior neck swelling that was not found to be associated with thyroid abnormalities. Tumor resection followed by radiotherapy was done, and paralysis of her left arm was noted afterward. Twelve years later, in 2013, she developed shortness of breath, for which she had a chest CT that showed a lobulated mediastinal mass, which was resected Six years later, in 2019, she noticed a left axillary mass, and postresection pathology results were consistent with neurofibroma, with immunohistochemistry results negative for keratin and CD34 but focally positive for S100.In June 2021, 3 months prior to her recent surgery, the patient complained of dyspnea on mild exertion, orthopnea, dysphagia to solids, and left arm pain and numbness. There was no history of weight loss, fever, or night sweats, and therefore, she underwent a chest CT with contrast, which showed a large lobulated hypodense mass in the left axillary region which involved suprascapular and clavicular components of about 7 × 10 × 15 cm. The mass was engulfing the major cervical vessels displacing the internal carotid arteries, elevating the left hemidiaphragm, and a necrotic supraclavicular lymph node was also noted at the base of the left side of the neck. In addition, an enhancing soft tissue mass on the right anterior chest wall indenting the second rib and widening neural foramina was seen .In September 2021, the patient was admitted on the basis of her last CT scans and having recurrent lesions extending to the mediastinum with a history of four prior resections in addition to her complaints of shortness of breath on mild exertion and orthopnea.The patient had a normal physical examination except for bilateral varicose veins and left upper arm paralysis. Multiple scars were noted on the neck and chest, indicating previous surgeries. Further examination revealed two left lateral cervical lesions, each measuring about 3 cm, in addition to a huge anterior lesion on the chest, extending to the left shoulder, causing loss of muscle mass and bone integrity, and associated with dilated superficial veins.Based on her history, physical examination, and prior CT scans, the patient was suspected of having a mediastinal neurofibroma and was scheduled for mediastinal mass resection, chest wall mass resection, and wedge resection of the left upper lobe of the lung.Chest CT and whole spine MRI showed extension of the tumor into the anterior upper mediastinum encasing major vessels including the aorta, brachiocephalic trunk, bilateral carotids, and subclavian arteries. The brachiocephalic veins were also severely compressed, with the left being completely obstructed, resulting in multiple collateral veins in the left chest wall and axilla. Bone erosion of the first and second ribs and the clavicles was noted. The enhancing soft tissue mass on the right anterior chest wall did not undergo any significant changes .Neck CT with and without contrast revealed a hypodense mass measuring 1.8 × 1 cm anterior to the left jugular vein, degenerative change in the cervical spine, and a few enlarged lymph nodes. Echocardiogram was normal, and the patient’s laboratory results were unremarkable.Mass resection surgery was performed under general anesthesia in a supine position with antiseptic scrubbing and covering of the skin. After anesthesia, the patient was prepared to be connected to a cardiopulmonary bypass machine in case of emergency. This was done by preparing the groin, inserting arterial and venous femoral access lines, and priming the machine. A sternothoracotomy incision (open book incision) was done. The mass was found to be adherent to the aortic arch, its branches, and the phrenic nerve. The aortic arch was seen after the mediastinal dissection of the mass. The tumor was carefully resected over the aortic arch from right to left. The tumor wrapped the brachiocephalic artery at its origin, and the dissection of the tumor in the area surrounding it was successfully done, resulting in the freeing of the artery after securing it using a vessel loop.The left common carotid and the left subclavian arteries were embedded in the tumor and therefore dissected and encircled using vessel loops. Resection of the tumor was done with both blunt digital and sharp scissors dissection.After separating the mediastinal mass from the chest wall and aortic arch vessels, the invaded part of the left upper lobe of the lung was stapled. A decision was made for staged surgery to remove the extrathoracic lesion in another session. Hemopneumostasis was done, and a chest drain was inserted. The wound was closed in layers, and the patient was then transferred to the ICU, where she was stable on mechanical ventilation.Follow-up CT scans revealed the extrathoracic parts of the tumor, and as of the publication date, the patient is scheduled for further surgery to resect them.The chest drains were removed on day 15 after the surgery, and the patient was discharged on day 20 postop in very well condition. Her medication list included acetylcysteine, furosemide, paracetamol, esomeprazole, and bisoprolol. Pathology results were consistent with a low-grade fibromyxoid sarcoma. By immunohistochemistry, tumor cells were diffusely positive for BCL2 and negative for CK7, S100, CD34, STAT6, MDM2, SMA, and beta-catenin. Grossly, the tumor cut surface was tan, white, and homogenous, with parts of it appearing mucoid. Microscopic analysis showed a biphasic tumor of mild to moderate to focally high cellularity set in fibrous and myxoid background. The tumor cells were mostly bland oval to stellate to spindle; focal areas showed increased nuclear atypia, and infarction-type necrosis was noted. Mitotic activity was not increased . An overThe tumor was resected in pieces, the three largest of which measured 16 × 15 × 2, 10 × 7.5 × 4, and 9 × 2.5 × 1 cm. Other smaller pieces measured between 1 and 4 cm.Fibromyxoid sarcomas represent 0.6% of all soft tissue sarcomas . LGFMSs On gross examination, LGFMSs can be as small in size as 1 cm or extend up to 20 cm –15. In oHistology-wise, LGFMSs are either infiltrative or circumscribed, with bland-looking spindle cells in a mixed fibromyxoid background. Most tumors typically show low to moderate cellularity, with mitotic figures being usually scant or undetectable. The myxoid areas of the tumor are usually found to have a curvilinear network of capillaries , 6.LGFMSs can present with a diverse range of unusual or high-grade histological features; some of them were present in our patient’s histological findings, most prominently, increased cellularity, nuclear atypia, and tumor necrosis. Other atypical features were not necessarily present in our case, including calcification, ossification, multinucleated giant cells, and other changes. Such features are not considered of prognostic value, nor have they been associated with tumor behavior , 6.Our patient’s condition was misdiagnosed as a neurofibroma until her last surgery. Neurofibromas are a common misdiagnosis for LGFMSs among other neoplasms due to similar histological features. Neurofibromas are benign tumors of the peripheral nerve sheath characterized by the random arrangement of their spindled cells in a myxoid background, in addition to the presence of thin wispy or thick bulky collagenous fibers . In contMUC4 gene is overexpressed in LGFMSs in addition to FOXL1 and CD34. MUC4, which is a high-molecular-weight transmembrane glycoprotein, remains the most sensitive marker for diagnosing LGFMSs . Some oten bloc resection, and compartmental resection were reported in the literature. Surgery is the only treatment that can result in disease-free periods (The mainstay of treatment of LGFMSs is surgical resection with clear margins. Other surgical approaches such as local excision, radical surgery, wide  periods , 18.This kind of surgery is very challenging but still doable. Surgeons can usually find the cleavage line and the proper plane for dissecting the tumor. The key to safe dissection in this patient’s case was to find the arch of the aorta, peel the tumor off it, and then continue upward in the same plane. The surgeon should be ready for the worst scenarios; the heart lung bypass machine should be ready and primed, femoral access should be prepared before the surgery, and lines for the cardiopulmonary bypass machine should be inserted before the operation.In our case, a staged surgical strategy was followed for two main reasons: the primary target of the surgery was to release the mediastinal compression from the heart and the great vessels, which was risking the patient’s life, and this was accomplished with a complex and very long operation. Dealing with the extrathoracic lesion in the same session would make it even more complex and prolonged. Another reason for this decision is the slow-growing nature of this tumor, making it possible to give the patient some time to recover with reasonable risk before undergoing any further surgical intervention.The use of chemotherapy and radiotherapy as a stand-alone or adjuvant treatment for LGFMS remains unclear, with many studies showing that it merely slows disease progression. Chemotherapeutic agent trabectedin has been shown to be effective in translocation-related soft tissue sarcomas. However, its role in the treatment of LGFMS remains unclear , 18, 19.Ideally, long-term management requires a rigorous follow-up involving whole-body scans. According to Maretty-Nielsen et al. , the besDespite their benign-appearing histology, low-grade fibromyxoid sarcomas are malignant tumors characterized by the high possibility of local recurrence in addition to metastasis, with most patients requiring multiple surgeries in their lifetime. This necessitates good diagnostic criteria to correctly identify the malignant lesions of LGFMS among other similar-presenting lesions that may direct the physician to follow a different course of treatment. Early detection and diagnosis lead to better outcomes with regard to complications and possible metastasis. Once diagnosed, proper and long-term follow-up using full-body scanning technologies, ideally using MRI, is required to detect any new lesions early in their course and improve outcomes. To our knowledge, no treatment protocol has been established for treating LGFMSs, and studies are still being conducted to determine the efficacy of chemotherapeutic agents and radiotherapies on these tumors. Because LGFMS can be somewhat challenging to diagnose in addition to being very rare, pathologists should be able to recognize the histology of LGFMS better and recommend the appropriate immunohistochemical tests to be certain of their final diagnosis.LGFMS patients suffer recurrent symptoms every few years, among which are compression symptoms caused by the size of the tumor and its malignant tendencies that may cause infiltration to other organs and therefore create an array of possible symptoms. LGFMSs’ tendency to recur requires patients to undergo multiple surgeries over the years, which might inflict on their quality of life, and take a mental toll on them and their families. Long-term follow-up might also be inconvenient for patients from a financial perspective and in terms of access and availability.


The current paper reports a rare case of gastric hyperplastic polyp in a dog with upper gastrointestinal hemorrhage, resulting in severe anemia. Gastric hyperplastic polyps are generally benign and found incidentally during endoscopic examination, or necropsy. Although they are asymptomatic, depending on their size or location, they can sometimes result in clinical signs including chronic occult blood loss, abdominal pain, or gastric tract obstruction. In human medicine, gastric hyperplastic polyps causing acute blood loss anemia have rarely been reported. However, there is no such case reported in veterinary medicine so far. To our knowledge, this is the first report describing a gastric hyperplastic polyp that caused severe anemia because of acute blood loss in a dog.An 11-year-old castrated male Shih Tzu was referred for lethargy and melena. The hematocrit level was 18.8% , indicating severe anemia. Abdominal ultrasound revealed a round-to-oval-shaped mass in the stomach. Computed tomography (CT) revealed an intraluminal mass (17 × 12 × 15 mm) cranial to the pyloric antrum. After obtaining informed consent from the owner, exploratory laparotomy and subsequent gastrostomy were performed, showing an ulcerated mass potentially responsible for the severe anemia. A lump of hair was firmly attached to the ulcerated surface of the mass. After complete removal of the mass, the anemia resolved spontaneously. Histological examination revealed that the mass was a gastric hyperplastic polyp. At the 6-month follow-up, the dog was healthy with a normal hematocrit level. Gastric hyperplastic polyps are tumor-like lesions arising from the mucosal surface of the stomach, and projecting into the lumen. They can appear in any part of the stomach, and are usually found incidentally during gastric endoscopy or necropsy. The clinical signs include chronic occult blood loss, abdominal pain, and gastric tract obstruction. Gastric polyps causing acute blood loss anemia have rarely been reported in human medicine. To our knowledge, this is the first report describing a gastric hyperplastic polyp that caused severe anemia because of acute blood loss in a dog. Gastric polyps are tumor-like lesions arising from the mucosa of the stomach and projecting into the gastric lumen, that can be either non-neoplastic or neoplastic lesions ,2,3,4. TGastric polyps can occur in any part of the stomach ,8. They There are several case reports describing gastric hyperplastic polyp causing acute bleeding in the human literature ,8,12. HoAn 11-year-old castrated male Shih Tzu was referred for lethargy and melena. On physical examination, the color of the mucous membrane was pale, and the capillary refill time was 2 s. A complete blood count revealed that the white blood cell count was 23,650/µL ; hematocrit (HCT) level, 18.8% ; hemoglobin level, 8.7 g/dL ; and platelet count, 424,000/µL . Blood chemistry analysis showed an alkaline phosphatase level of 275 U/L . Blood samples were collected and submitted for the anemia panel, which showed negative findings.Thoracic and abdominal radiography revealed no significant findings. Abdominal ultrasonographic examination  revealed a solitary and round-to-oval-shaped intraluminal mass cranial to the pyloric antrum . In ordeComputed tomography  was performed to evaluate the gastric lesion. With the patient positioned in ventral recumbency, a contrast study was performed after administration of the contrast medium . Contrast uptake was assessed in the late venous phase. Post-contrast CT images revealed a mass (17 × 12 × 15 mm) cranial to the pyloric antrum, and the contrast enhancement of the mass was similar to that of the surrounding gastric wall . No contEndoscopy  was performed to further observe gross appearance of the mass in the stomach. The mass was located cranially to the pyloric region A.Immediate blood transfusion was performed at the time of presentation, and the HCT level subsequently increased to 35.3%. However, the HCT level decreased to 28.7% two days after blood transfusion. Although bleeding was not identified, exploratory laparotomy was performed with informed consent from the owner.Preanesthetic agents, including butorphanol  and midazolam , were administered intravenously. After administration of propofol , inhalation anesthesia using isoflurane  was maintained during surgery.With the patient in dorsal recumbency, the skin and abdominal wall were incised routinely. The mass within the stomach was palpated cranial to the pyloric antrum. Gastrostomy was subsequently performed, and the mass was identified on the dorsal wall of the gastric body. No source of bleeding was found at the cranial surface of the mass, as identified during endoscopy. However, in contrast to the cranial surface of the mass, the caudal surface of the mass was ulcerated with hemorrhage, which was considered to be a bleeding focus. Moreover, a lump of hair was firmly attached to the mass B. The maThe hematocrit level of the patient increased spontaneously, reaching 32.9% on the ninth day after surgery, and 35.5% on the twelfth day. The dog was discharged on the twelfth day after surgery. At the 2-month follow-up, the dog showed unremarkable recovery. At the 6-month follow-up, the hematocrit level was 44.0%.Histologic examination  revealedHelicobacter (H) pylori in more than 90% of patients [H. pylori eradication might be attributed to complete resolution or significant size reduction, although the exact role of H. pylori in the formation of gastric polyps is unclear [H. pylori, revealed a negative result. Gastric hyperplastic polyps occur secondary to inflammatory responses, such as gastritis, and result from an exaggerated mucosal response to injury [Gastric polyps are defined as lesions protruding from the mucosal layer into the lumen of the stomach . These ppatients . Thus, s unclear ,10. In to injury . Since tGastric polyps are usually asymptomatic. However, depending on their size or location, they can result in clinical signs, including vomiting or intermittent gastrointestinal obstruction . Antral Biologically, gastric polyps are classified as neoplastic or non-neoplastic. For domestic animals, the World Health Organization classifies non-neoplastic polypoid growth into two entities: hyperplastic, and inflammatory polyps. Gastric hyperplastic polyps are typically observed in the antrum of the stomach. Histologically, they consist of hyperplastic, elongated, and distorted pits extending deep into the stoma and containing pyloric glands, chief cells, or parietal cells. On the other hand, inflammatory polyps are characterized by the presence of a normal epithelium lining a granulation tissue infiltrated by mixed inflammatory cells, including lymphocytes, or plasma cells. They are also sometimes accompanied by lymphoid aggregates with germinal cores ,8,11.Studies describing cases in human patients suggested that atypical surface morphology and bleeding in gastric hyperplastic polyps may be associated with malignant changes . HoweverSurgical treatment depends on the type and location of the polyps in the stomach . EndoscoIn conclusion, the significance of gastric hyperplastic polyps in gastrointestinal blood loss has not been reported in veterinary medicine. If the gastric polyp is suspected to be the only reason for severe upper gastrointestinal hemorrhage, gastric hyperplastic polyps should be considered in the differential diagnosis of cases presenting with severe anemia.


Phosphaturic mesenchymal tumors (PMT) are a rare neoplasm oftentimes associated with tumor-induced osteomalacia (TIO). The non-specific presentation and symptoms of these pathologies make them difficult to diagnose. We report a case of a 52-year-old patient with an intermetatarsal phosphaturic mesenchymal tumor who presented to the orthopedic sports medicine clinic with metabolic deficiencies and bilateral subtrochanteric cortical stress fractures indicative of osteomalacia. The tumor was entirely resected within nine months of symptom onset and has shown no recurrence at the one-year follow-up. This case report characterizes an unusual cause of stress fractures presenting to orthopedic sports medicine clinics and the variability in the presentation of phosphaturic mesenchymal tumors. Phosphaturic mesenchymal tumors (PMT) are a rare neoplasm associated with tumor-induced osteomalacia (TIO) . MechaniThe non-specific symptoms of TIO and associated PMTs make them difficult to diagnose. Retrospective studies have found the average time between symptom presentation and surgical tumor resection to be greater than five years ,5. DespiTo help physicians who treat metabolic bone disease and stress fractures diagnose and treat this rare disease, we report a case of a non-malignant PMT between the second and third metatarsal of the left foot with TIO. The patient was informed that data concerning the case would be submitted for publication and provided his consent.A 52-year-old man presented to an outpatient clinic with intermittent, sharp pains upon hip flexion. He was seen by a physician’s assistant and referred to orthopedics sports medicine. He presented to the sports medicine physician with antalgic gait and abnormal internal rotation of the left hip but normal hip strength. An X-ray of the pelvis and hip showed proximal femoral shaft lucencies that were incomplete fractures indicative of osteomalacia Figure . In a foAt the one-month follow-up with the sports medicine physician, his pain had spread bilaterally to both hips and he was walking with a cane. The vitamin D supplementation returned levels to 36 ug/L but the radiographic scans showed no healing of the fractures. Four months later, the subtrochanteric cortical fractures remained bilaterally and a dual-energy X-ray absorptiometry (DEXA) scan was ordered. The DEXA scan revealed a T-score of -2.1 in the left hip, indicating an increase in fracture risk due to osteopenia  1,4,7,10-tetraazacyclododecane-1,4,7,10-tetraacetic acid (DOTA)-octreotate  positron emission tomography (PET)/computed tomography (CT), a newer modality to detect neuroendocrine tumors, can detect up to 100% of tumors . HoweverA biopsy of the tumor in our patient showed characteristic histopathological and immunohistochemical features of a PMT. The PMT displayed bland, spindled, and stellate shape cells embedded in a smudgy basophilic matrix. Other common features seen in PMTs include cells with vesicular chromatin and a sheet-like arrangement . The tumDue to the variability and rarity of PMTs, early diagnosis is oftentimes difficult. However, the majority of PMTs are benign, allowing marginal resection to be a curative treatment for TIO ,18. IncrPhosphaturic mesenchymal tumors and associated tumor-induced osteomalacia can be an unusual cause of stress fractures. Presentation of these tumors can be highly variable and may present to the sports medicine clinic. In cases of unusual stress fractures, patients should be asked about the presence of soft-tissue masses. When osteomalacia is suspected, especially in cases without a clear underlying cause, detection of elevated FGF23 and hypophosphatemia are indicators of possible tumor-induced osteomalacia and should prompt further diagnostic workup to identify an underlying tumor.


Malignant inflammatory myofibroblastic tumors (IMT) are extremely rare, aggressive tumors with variable presentation. This is a case of a 29-year-old female presented with severe anemia and a large abdominal mass presumed to be a gastrointestinal stromal tumor (GIST). Severe anemia, leukocytosis and thrombocytosis accompanied the presentation. Final pathological assessment yielded a diagnosis of malignant IMT. Given the rarity of these tumors, no established diagnostic criteria exist aside from histological analysis of the tissue, which may result in delays or inappropriate treatment. As these tumors are aggressive in nature, a high index of suspicion is critical to improve outcomes. Further reports on the presentation, diagnosis and treatment of such rare tumors are important to develop clinical diagnostic guidelines to improve diagnosis and treatment and improve outcomes. Inflammatory myofibroblastic tumors (IMT) are uncommon tumors first described in 1939 . DiagnosA 29-year-old female presented to hospital with abdominal pain. She had anemia (~110 g/L) and intermittent low-grade leukocytosis. She had been having persistent abdominal pain, intermittent nausea and drenching night sweats. She denied weight loss, fevers and reported no other notable symptoms. Her past medical history was unremarkable and past surgical history included two caesarean sections. She was not taking any medications and had no allergies. She was a non-smoker and social alcohol user. On examination, she was febrile with a temperature of 38.1°C, but all other vital signs were within normal limits. Her abdomen was tender to palpation, and there was a palpable mass in the epigastric region.Investigations revealed an elevated WBC count of 35 × 10e9/L, a hemoglobin of 72 g/L and a CRP of 141 mg/L. A CT scan revealed a 15 × 11 cm multi-lobulated, heterogeneous, necrotic, exophytic mass arising from the stomach as well as multiple peritoneal deposits and five suspicious masses in the liver . Upper eOne month later, she re-presented with symptomatic anemia (hemoglobin of 56 g/L). Her leukocytosis had worsened, ranging from 38–44 × 10e9/L. She was urgently referred to a surgical oncologist for definitive management. Her biopsies reported a spindle cell neoplasm—CD117 and DOG-1 negative. However, given the anemia, the heterogeneous/ulcerated appearance on imaging, and the presence of liver metastases, a preliminary diagnosis of a GIST was made. After discussion at multidisciplinary tumor board rounds, and given her ongoing bleeding, the decision was made to proceed with upfront surgery. She underwent a subtotal gastrectomy, transverse colectomy, distal pancreatectomy, splenectomy, drainage of intraabdominal abscess and formation of an end colostomy. Intraoperative findings demonstrated infiltration of the mass into the transverse colon with perforation into the retroperitoneum. There were multiple omental nodules. Final pathology reported a negative gastric resection margin.Postoperatively, her hemoglobin and leukocytosis improved (hemoglobin of 95 g/L and a WBC of 20 × 10e9/L); however, she developed a large pulmonary embolus and was placed on anticoagulation. A one-month follow-up CT scan showed progression of disease with a gastric mass measuring 6 cm, multiple enlarged mesenteric masses and enlargement of the hepatic metastases . Final pIn follow-up, she continued to have a gradually increasing WBC count reaching as high as 205 × 10e9/L and a persistent anemia despite ongoing transfusions. Another CT scan was performed showing significant progression of multiple large soft tissue masses in the abdomen as well as sclerotic osseous metastasis. Given the progression of disease, she was switched over from Crizotinib to Alectinib. Her disease continued to progress and 4 months following her surgery the patient passed away from her disease.IMT is a spindle cell tumor containing multiple inflammatory cells . It has IMTs are diagnosed histologically; however, histological characteristics do not provide any indication on prognosis . ActivatAnother unique quality that has been reported in cases of IMT is patients presenting with laboratory abnormalities such as anemia, leukocytosis and elevated platelet count , 9, 10. Although there is a limited body of literature surrounding IMT, there are reports detailing a tendency for local recurrence, but with a small risk of distant metastasis . As demoIMT are locally aggressive tumors affecting predominantly young adults. The malignant variety including distant metastatic disease is rare. The cornerstone of treatment is a complete surgical resection but has evolved to include ALK targeted therapy. Rates of local recurrence are high (approximately 25%), and it remains a challenging disease to manage. Given the vague history in the current, albeit limited body of literature, increased reporting of these rare malignancies is needed to assist in diagnosing and treating it. As more case reports become available, we can begin to develop diagnostic, treatment, and follow-up guidelines to improve management and patient outcomes.Written informed consent was obtained from the husband of the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.No conflict of interest to disclose.This research did not receive any specific grant from funding agencies in the public, commercial or not-for-profit sectors.Provenance and peer review. Not commissioned, externally peer-reviewed.


A 27-year-old woman was confirmed to have extraabdominal desmoid-type fibromatosis. Desmoid-type fibromatosis is a very rare connective tissue neoplasm with the extraabdominal type even more rare. It is most commonly found in proximal structures such as shoulders, neck, chest, and extremities. There are few case reports for desmoid-type fibromatosis located in paraspinal soft tissue. We report a case of desmoid-type fibromatosis mimicking a myxofibrosarcoma.Teaching Point: Even if there are suggestive findings for malignant soft tissue tumor on radiologic evaluation, histological confirmation is necessary before surgical treatment. Extraabdominal desmoid-type fibromatosis (DF) is a very rare condition that arises from the musculoaponeurotic structures 2. AlthouThe purpose of this case report is to inform that it is necessary to confirm the diagnosis by preoperative biopsy, even if there are imaging findings highly suggestive for malignancy.A 27-year-old woman presented with localized swelling and back pain for seven months. She had a history that included a traffic accident 21 months ago and had immediate T5 and T6 corpectomy with posterior fixation. Clinical examination revealed localized tenderness adjacent to the lower part of the operation scar on the back. The patient had no neurologic symptom or specific familial history. Contrast-enhanced computed tomography (CT) revealed a heterogeneously enhancing mass, located in the left posterior paraspinal soft tissue . MagnetiDF is a locally aggressive fibroblast and myofibroblast neoplasm, usually found in the deep intermuscular spaces of soft tissue. In addition, DF shows an invasive growth pattern and tends to have considerable rate of local recurrence after treatment . PatientMRI is the most reliable technique for diagnosing DF which shows iso- to mildly hyperintense relative to muscle on T1-weighted images, and iso- to hyperintense relative to muscle on T2-weighted images with inner band-like areas showing low signal intensity (SI). However, because of the heterogeneity of the lesion, malignancy cannot be excluded on imaging. In our case, the large sized mass located in subfascial area of back muscles with T2 bright high SI and intense contrast enhancement suggested a myxoid component. Furthermore, the fibrotic band and peripheral tail sign, which are well-recognized prognostic factors of soft tissue sarcoma , were alFor treatment of extraabdominal DF, surgical excision and/or radiation therapy can be considered. Hormonal therapy, such as tamoxifen or progesterone, or chemotherapy also may be considered for not easily resectable lesions . Neverth156Although clinical and radiological evaluations suggest malignancy, histological confirmation before active treatment is essential.


Paraneoplastic SIADH has been well documented with small cell carcinoma of the lung. The following case report describes refractory hyponatremia due to SIADH associated with an immature teratoma. Complete resolution occurred following surgical debulking.2A 27-year-old previously healthy gravida 1, para 1, presented with a three month history of worsening diffuse abdominal discomfort, nausea, and vomiting. She also reported a one week of fatigue, dizziness, blurred vision, and a mild headache. She denied syncope, seizure-like activity, urinary changes, and abnormal vaginal bleeding. Her exam was notable for a large abdominopelvic mass and no clinical signs of fluid overload nor edema. On contrast-enhanced CT scan, there was a 20-centimeter complex mass with associated ascites, peritoneal thickening, omental infiltration, and borderline retroperitoneal lymphadenopathy. Tumor markers were elevated with a CA-125 of 191 U/mL and an AFP of 69.3 ng/mL. Her LDH was normal at 150 U/L and her quantative HCG was <1 mIU/mL. On pre-operative labs, she was found to have a serum sodium of 116 mmol/L. Her liver enzymes and her other electrolytes were within normal limits.She was admitted for further hyponatremia work-up and management. Orthostatic blood pressures were obtained and were consistent with euvolemia. Admission labs were consistent with SIADH with the following significant results: urine chloride was 162 mmol/L, urine sodium was 229 mmol/L, and urine osmolality was 891 mOsm/kg. Her thyroid stimulating hormone and morning cortisol were within normal limits. Upon admission, she initially received intravenous fluid resuscitation due to concerns for diarrhea associated hypovolemic hyponatremia. This intervention worsened her hyponatremia, further suggesting the diagnosis of SIADH. As such, her management then included strict fluid restriction to 1.5 L daily, 1 g salt tabs three times daily, and intermittent furosemide infusions. Her salt tabs were increased to 2 g three times daily and her furosemide was changed to scheduled 20 mg twice daily after minimal improvement. After 5 days there was a modest improvement of the serum sodium but hyponatremia persisted.During this admission, she became febrile to 101.9 degrees Fahrenheit. A full sepsis work-up was negative aside from the detection of enteropathogenic E. Coli (EPEC) on stool testing. This finding was consistent with her reported history of intermittent diarrhea, and she was treated with a 7-day course of antibiotics.She ultimately underwent an exploratory laparotomy with resection of the 20-centimeter tumor involving the right ovary and fallopian tube. Intraoperative findings also included 3 L of ascites with multiple 2 mm nodules in the omentum and pelvic cul-du-sac, which were removed. She was repleted with 500 ml of albumin and 1 L of normal saline. The intraoperative rapid frozen section pathologic evaluation of the tumor was inconclusive, and given the patient’s desire for preservation of fertility, the decision was made to resect the peritoneal nodules and omentum. There were no complications from her surgery.The final pathology demonstrated stage 3B grade 2 immature teratoma arising from the right ovary. On postoperative day 1, her serum sodium normalized and remained within normal limits throughout her hospitalization as demonstrated in 3Pathologic examination of the specimen revealed immature teratoma elements involving the right ovary, omentum, and peritoneal nodules. The ovarian tumor measured 20 cm in greatest dimension and weighed 1704.4 g. The cut section of the bivalved tumor demonstrated an irregular, predominantly solid, sporadically cystic, yellowish mass A. Micros4This patient presented with classic symptoms of hyponatremia: nausea, headache, and fatigue. Having a normal thyroid stimulating hormone level makes the diagnosis of hypothyroid-related hyponatremia less likely. She was not taking medications at the time of her diagnosis. The patient did have malignant ascites as well as diarrhea, which could lead to hypotonic hyponatremia; however, she did not respond to fluid resuscitation. The patient also had an inappropriately high urine sodium level, which commonly is associated with SIADH. She partially responded to medical management, water restriction, and salt tablets; however, the serum sodium did not normalize until the immature teratoma was resected.One of the initial cases of refractory hyponatremia was associated with a serous ovarian carcinoma with neuroendocrine differentiation . There h5Malignancy associated syndrome of inappropriate antidiuretic hormone secretion is rare. Here, we describe a case of immature teratoma associated SIADH which was refractory to medical therapy and only resolved following surgical debulking. Hyponatremia due to paraneoplastic syndrome is rare, but should be considered in the diagnosis differential of a young woman with a pelvic mass and refractory hyponatremia.6Informed consent to write the case report was obtained from the patient.All of the listed authors in this manuscript were an active part of the surgical and medical treatment of the patient, as well as contributed to the writing of the manuscript.The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.


Lymphangioma is a localized lymphatic malformation, that occurs more frequently in the head and neck. Its etiology remains unknown; some authors believe it consists of a congenital malformation of the lymphatic vessels, while others believe that some acquired factors lead to lymphatic obstruction, lymph retention, lymphangiectasia and proliferation. Its incidence is 1 case for 6000–16,000 live births.It can occur at any age, but approximately 50% of cases are present at birth and 90% are diagnosed by 2 years of age. Both genders are equally affected. The most common affected sites are lips, tongue and neck. Infection, trauma and bleeding can trigger rapid growth of the lesion.We report the case of a child diagnosed with submucosal microcystic lymphangioma with pharyngeal lesions that required tracheostomy due to upper airway obstruction. Treatment is described with OK-432 that allowed subsequent decannulation.A two-month old male patient was brought to the emergency department of a tertiary hospital in southern Brazil with stridor and respiratory effort. He was submitted to airway endoscopy under general anesthesia, which disclosed pharyngeal mucosa infiltration, from the nasopharynx to the hypopharynx, with upper airway obstruction .Figure 13) located in the retropharyngeal space, on the right  showed a massive multiloculated lesion , microcystic (more common in the tongue and cheek) and mixed. Spontaneous regression occurs in 1.6–16% of cases, with regression commonly being followed by recurrence.The diagnosis is made through clinical presentation and imaging tests, such as cervical ultrasound and MRI, which define the size and extent of the disease.The main goal of treatment is to restore function preservation and esthetic integrity.In some mildly symptomatic cases there are reports of lesion involution in 15%–70% of cases; such children can be followed until the age of five years.Surgical resection is the definitive treatment and it is indicated in lesions larger than 3 cm, with progressive growth, bone erosion, dyspnea, dysphagia or esthetic deformity.The surgical mortality rate can be up to 6%, and the rate of complications ranges from 19% to 33%. The recurrence rate is up to 53%.Other current treatments include radiation therapy, laser, and sclerotherapy with OK-432, doxycycline, sodium morrhuate, dextrose, tetracycline, hypertonic saline solution, acetic acid, ethanol, cyclophosphamide, interferon, fibrin glue, corticosteroidsRadiotherapy may be indicated in recurrent and persistent cases that remain symptomatic or as an adjuvant to surgery. However, there is a risk of malignancy development, and a series of cases showed it was not an effective treatment.2 or YAG, allows a more accurate lesion removal, without functional impairment.Endoscopic laser resection, either with COThe 95% or 100% alcohol solution is the most potent sclerosing agent, but can damage the vascular endothelium and denature the proteins and cause thrombosis. Adverse effects include nerve damage, pain, edema, systemic symptoms. The safe amount to be used in young children with large lesions is limited and often inefficient.Bleomycin is a cytotoxic antibiotic that induces degradation of deoxyribonucleic acid, causing lesion sclerosing and fibrosing.Doxycycline is a broad-spectrum antibiotic that cause fibrosis and prevents lesion cell proliferation. Due to the discomfort of the injection, it should be applied under general anesthesia and pain-control medication are needed after the procedure.The injection of corticoids, such as triamcinolone,Few authors have reported on the use of fibrin glue, which would act as a hemostatic agent and would eliminate the dead spaces in the cysts; no side effects have been reported.Interferon-alpha 2a has shown to be effective in a small group of children, but it is not extensively used due to its adverse effects: fever, nausea, diarrhea, weight loss, liver enzyme abnormalities, alopecia, neutropenia, headaches.The intralesional use of cyclophosphamide was described in a study on life-threatening tumors. The most common adverse effects are: bone marrow suppression, hemorrhagic cystitis, liver dysfunction and long-term malignant transformation.Streptococcus pyogenes strains treated with benzathine penicillin. After its injection into the lymphangioma, it stimulates the proliferation of lymphatic endothelial cells and obstruction of lymphatic channels. Its concentration is 0.1 mg/10 mL. The dose should not exceed 20 mLOK-432 or picibanil is a biological preparation containing OK-432 leads to complete response in macrocystic lesions, with a lower degree of response in mixed or microcystic lesions.If the lesion does regress within 90 days, a new application with up to 20 mL can be performed.Several reports have concluded that OK-432 is safe and effective for the treatment of lymphangiomasThere are few reports about the effects of treatment with OK-432 in microcystic submucosal lymphangiomas in patients who required tracheostomy. There is little evidence on each therapeutic measure and most of them are based on case reports or series of cases. This is the first report on the use of this medication in this type of lesion that allowed patient decannulation.The authors declare no conflicts of interest.


A 58‐year‐old right‐handed man presented to our tertiary care center with gliosarcoma (GS) infiltration through the dura, skull, and soft tissue. Patient had a previous history of right temporal GS, with four intracranial surgeries prior to presentation. A multidisciplinary approach was used to treat the lesion and perform reconstruction. It is important to consider a multidisciplinary approach when treating highly invasive tumors such as GS and to be mindful of mechanisms of spread while planning resection in index surgeries. Initially, he presented to an outside hospital with seizures. That hospital performed index resection in June 2020, and he underwent adjuvant radiation and temozolomide at the same institution. His index resection was at the outside hospital in June 2020, and he underwent adjuvant radiation and temozolomide at the same institution. He had two further resections following this surgery. The second surgery was in October 2020; it was complicated by wound infection requiring washout at which time native bone was left in place. The patient also was found to have bony metastasis in the cervical spine for which he underwent corpectomy and instrumentation in January 2021 prior to a third debulking in February 2021.The patient was initially evaluated by video telecommunication by our team in April 2021; his chief complaint during his virtual visit was scalp firmness and pain. He was admitted to our center shortly thereafter for in‐person evaluation and surgical planning. The otolaryngology/head and neck surgical service evaluated the patient in‐house for planning. Magnetic resonance imaging (MRI) and venogram (MRV) were obtained. Compared to previous imaging, there was substantial tumor recurrence within the resection cavity and peri‐resection bed invasion, as well as enhancement within dura and surrounding bone including petrous temporal, parietal, and occipital bones, and soft tissue of the scalp, which correlates to the gross findings seen on physical examination Figure .Two days after admission, after appropriate imaging was obtained, the patient was taken for operative management. Management was multidisciplinary, with different specialties collaborating on operative treatment. First, the head and neck team outlined and performed a wide local excision on a 14‐cm section of scalp that grossly appeared to have tumoral invasion Figure . The supPostoperatively, a lumbar drain was placed for 72 h of CSF diversion for flap protection as well as CSF leak prophylaxis, as the temporal bone was significantly resected. The patient had an unremarkable postoperative course and was discharged to skilled nursing facility. Final pathology showed gliosarcoma invasion in scalp, dura, and bone. Scalp resection was extensively sampled with evidence that radial margins were free of tumor.3Given the rarity of gliosarcomas with respect to central nervous system tumors, much of the literature describing their diagnosis, treatment, and management is limited to case reports and small retrospective studies. The case description above demonstrates a unique presentation of gliosarcoma—one that involves extracranial presentation by way of direct extension through the dura mater, skull, and soft tissues, distant metastases, as well as a treatment strategy for addressing the extracranial extension.Currently, the World Health Organization classification system includes gliosarcoma as a subtype of GBM. Limited literature suggests, however, that gliosarcoma exhibits features, which may warrant clinical distinction including histologic and radiologic appearance, as well as its propensity to metastasize. Case reports and small series have reported metastases to the lung, liver, cervical lymph nodes, spleen, adrenal glands, bone marrow, kidneys, oral mucosa, skin, skull, ribs, and spine.As a direct consequence of the lack of treatment and outcome data for GS, and its current classification as a glioblastoma subtype, the treatment of gliosarcoma exactly mirrors that of GBM. In the present case, that treatment paradigm was followed upon initial presentation to the outside hospital, including an attempt at gross total resection followed by adjuvant radiation and temozolomide. However, when combining this case's ultimate metastatic presentation at our institution, similar case series describing metastatic presentations after index surgeries, and the known propensity of the sarcomatous portion to metastasize, attempt at surgical resection may have to be weighed against the risk of metastasis. This thought process may be expounded upon when considering the recent development of targeted molecular therapeutics in the treatment cancer, including PIK3 inhibitors  and PTEN inhibitor, two genes found to have a high prevalence of mutation in sequenced tumor samples.Collin M. Labak, M.D participated in surgery, assisted with drafting paper. Nicholas M. Rabah, Jasmine P. Kipke, Uma V. Mahajan, and Kyle B. Labak assisted with drafting and revisions. S. Ahmed Ali, M.D, Nicole Fowler, M.D, and Andrew E. Sloan, M.D performed surgery, reviewed manuscript.The authors have no conflicts of interest to report at this time.Written informed consent was obtained from the patient to publish this report, in accordance with the journal's patient consent policy.


Cystadenoma and cystadenocarcinoma of the biliary duct remain a rare diagnosis, the localization is intrahepatic in 97% of the cases and extrahepatic in 3%of them [1]. Cystadenocarcinoma of the gallbladder is the rarest localization, it's usually diagnosed late because of the poor symptoms and the nonspecific lesions in the different imaging.Only hundreds of cases have been reported for cystadenocarcinoma of the gallbladder.This case report has been reported in line with the SCARE Criteria.We present a 70-year-old woman, with no family or personal history, who was complaining of pain in the right hypochondrium and an altered general well-being, with no other clinical abnormalities.The imaging including abdominal sonography, CT scan, and MRI concluded at a budding lesion formed in the gallbladder wall which measured 65 × 15mm.Cancer of the gallbladder was suspected and the patient was operated. A resection of segment s4b and 5 of the liver was performed with lymphadenectomy. The post-operative clinical course was uneventful.The diagnosis of cystadenocarcinoma of the gallbladder was confirmed on an anatomopathological exam of the specimen.Cystadenocarcinoma is a rare diagnosis.There are no specific symptoms or lesions at the imaging.The confirmation is obtained on an anatomopathological study of the specimen. •Cystadenocarcinoma is a rare diagnosis.•There are no specific symptoms nor lesions at the imaging.•The confirmation is done on an anatomopathological study on the specimen. The first case to be reported was by EDMONSON in 1958 [It is usually observed in the intra or extrahepatic bile ducts , and the2A 70-year-old woman, with no family or personal history, was complaining of pain in the right hypochondrium, and an altered general well-being with no other symptoms.Examination showed tenderness in the right hypochondrium, with no jaundice and no palpable abdominal mass.Hepatic blood tests were normal. Carcinoembryonic antigen (CEA) and Carbohydrate Antigen (CA19-9) were normal. Alpha-fetoprotein (AFP) was above the superior limit.The abdominal Sonography showed a thick-walled hyperechoic lesion with lithiasis of the gallbladder.The CT scan showed a bifocal hypodense lesion in the gallbladder wall Fig. 1ArtThe MRI showed a bi-focal lesion developed on the wall of the gallbladder that measured 65 × 15mm associated with multiple lithiasis .Fig. 2BiA cancer of the gallbladder was suspected and we opted for a surgical treatment.A bi-segmentectomy s4b and s5 of the liver with extended lymphadenectomy were performed by a professor in general surgery in la Rabta hospital in Tunisia .Fig. 3BiThe diagnosis of cystadenocarcinoma of the gallbladder was confirmed on an anatomopathological exam of the specimen classified as pT2aN0 with harvested lymph nodes of 15, all being reactive.The post-operative clinical course was uneventful. The patient noted an improvement in her general wellbeing.The follow-up based on multiple CT scans and tumor markers was regularly conducted until august 2022. It showed no recurrence of the tumor.3Cystadenocarcinoma of the biliary duct is usually observed in women, at the age range of 40–60 years Old. In a histopathologic study of 540 consecutive cholecystectomies, Terada  reportedIt has no specific symptoms. The most common complaint by the patients is right hypochondoralgia or bloating.Examination usually shows tenderness in the right hypochondrium, sometimes a palpable mass in the upper abdomen or even jaundice.CA19-9 and ACE are not specific for cystadenocarcinoma. Sang  reportedUltrasonography is the first imaging indicated. Lesions may vary and are not specific for cystadenocarcinoma. We essentially observe a thick-walled lesion developed in the gallbladder as observed in our case. Sometimes we find a cystic lesion with many septations and lymph nodes.The MRI is more specific to define benign and malignant lesions .A fine needle aspiration for cytological study must be avoided to prevent the spreading of malignant cells . The conAs a malignant lesion is suspected, we perform a radical surgery based on a liver resection s4b and 5 with extensive lymphadenectomy.Even though the prognosis of our patient is prosperous and the surgical treatment was radical, recurrence may be occurred in 15% of the cases [A regular follow up with clinical examination, tumor markers and CT scan for 5 years is advised.4Cystadenocarcinoma of the biliary duct is rare, mostly intrahepatic.The extra hepatic lesions including the gallbladder, like the one we represent in our case report represent 3% of all cases combined.The symptoms are poor and the tumor markers aren't specific. The lesions at the imaging only show thickening off the wall, or a cystic lesion with multiples septations.Bisegmentectmy of the liver s4b and 5 with lymph node resection is a complete and sufficient treatment.The diagnosis of cystadenocarcinoma of the liver is made on an anatomopathological exam of the specimen.Locoregional or distant recurrence is observed in 15% of the cases even after complete resection with margin-negative surgery.Not applicable.No sources of funding.Hammami yasmine, Conceptualisation, Redaction, Data curation, Project administration/Sebai amine, Conceptualisation, Redaction, Data curation, Project administration/Ouadi Yacine Conceptualisation, Redaction,/Ben Mahmoud Ahmed Photography rendering/Frikha Wassim Photography rendering, Data curation/Bel haj Ali jihene Data curation/Fterich Fadhel Samir Supervision, Validation, Visualisation/Montasser Kacem Supervision, Validation, Visualisation.Name of the registry:Unique Identifying number or registration ID:Hyperlink to your specific registration (must be publicly accessible and will be checked):Hammami Yasmine.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.Not commissioned, externally pee-reviewed.All authors declare that they have no any conflicts of interest.


Extensive loss of soft tissue and bone due to neoplasia, trauma, or infection in extremities often leads to amputation.We present the case of a 72-year-old female patient presenting with an extended cutaneous squamous cell carcinoma of the lower leg, developed on top of necrobiosis lipoidica. After achieving the R0 resection, a 26 × 20-cm soft tissue and 15-cm tibial bone defect resulted. The contralateral leg had been lost due to the same disease 18 years before. We achieved a successful reconstruction of the leg using a pedicled fibula transplantation, an extended anterolateral thigh perforator flap, and an internal fixation with plate and screws. Two years after the original surgery, the patient is relapse-free and mobile, with adequate function of the reconstructed foot.Our case presented a unique combination of pedicled fibula transplantation and free extended ALT perforator flap to reconstruct an extensive defect after resection of a rare cSCC on top of NL. In selected cases, the boundaries of limb salvage can be pushed far beyond the current standards of treatment.The online version contains supplementary material available at 10.1186/s12957-023-02923-z. Cutaneous squamous cell carcinoma (cSCC) is the second most common cancer in humans. It can agressively infiltrate local tissues and has a reported recurrence rate of 2.7 to 4.6% and a rate of metastasis of 1.2 to 4%. The locally spread disease can be treated by surgery, radiotherapy, or combined treatment. In cases of advanced local disease, anti-PD1 therapy can be considered [Large bony defects of the tibia can be reconstructed by the Masquelet technique, by bone transport, or by vascularized bone transplantation . While tThe free anterolateral thigh (ALT) flap has become over the years a workhorse flap in reconstructive surgery. With a reliable blood supply, it can be harvested as an extended flap to cover extremely large soft tissue defects .Bacteroides fragilis could be isolated and the antibiotics were adapted to the antibiogram. At the next surgery, further debridement and lavage with the exchange of the plate and screws was performed. The remaining soft tissue defect was covered with a second ALT flap measuring 16 × 7 cm from the left thigh, including the fresh split skin donor sites, using two perforator vessels (Fig. We hereby present the case of a 72-year-old woman with an extended cSCC of the leg with tibial infiltration, developed on a chronic ulcer that developed on top of NL, without diabetes mellitus Fig. a–d. EighOne year postoperatively, the CT scan showed insufficient bony consolidation at the proximal and distal tibia-fibula transition despite exogenic ultrasound therapy, so a revisional surgery was performed. The extended ALT flap was longitudinally split to provide access to the plate while avoiding the lesion of the vascular pedicle. Two osteosynthesis plates were used to stabilize the leg medially and laterally and to ensure bone consolidation. Autologous cancellous bone was applied at the transition between the fibula and tibia proximally and distally. Postoperatively, the wounds healed without issues. After 6 weeks, mobilization of the leg was started using a fitted boot with pressure distribution to the tibial tuberosity and the foot. After 3 months postoperatively, the exogenic ultrasound therapy was continued for another 3 months. The pressure distribution was progressively switched from the tibial tuberosity to the foot.Two years after the beginning of treatment, the boot was completely removed. The patient was mobile on the reconstructed leg and below-the-knee prosthesis on the contralateral side Fig. a,b. The In this case report, we present the successful treatment of a very complex bone and soft tissue defect of the lower leg using innovative microsurgical reconstructive techniques.NL is a rare granulomatous disease, which in very rare cases may degenerate into cSCC. A literature review by de Souza et al. identified only 16 published cSCCs on top of NL . AlthougEven if surgery is the first-choice treatment for cSCC, immune checkpoint inhibitors gained a leading role in the treatment of locally advanced and metastatic cSCCs. In our case, surgical therapy was preferred because the tumor was still resectable and an anti-PD1 therapy failure could not be ruled out in a cSCC arising in NL .After R0 resection, a 15-cm tibial defect resulted. This defect could have been reconstructed using bone transport or the induced membrane technique described by Masquelet. Although technically feasible, we preferred due to the age of the patient and the dimension of the bony defect an interposition with vascularized bone to avoid complications like bone exposure or bone resorption. The free vascularized transfer of the contralateral fibula is a well-known procedure and ensures a fast interposition of the tibial defect, with the price of supplemental donor site morbidity in the contralateral, usually healthy leg. In our case, this procedure was rendered impossible by the existing lower leg amputation. As the ipsilateral fibula was healthy and not affected by the tumor resection, this appeared to be the fastest and safest solution for the patient. The salvage of the remaining leg was of utter importance for our 72-year-old patient. With a three-vessel blood supply of the leg, the fibula could be skeletonized with the fibular artery and veins as pedicle. The blood supply of the remaining musculature from the anterior and lateral compartments was secured by the anterior tibial artery and veins. Khira et al. reported over 12 patients who received a reconstruction of the tibial shaft with a pedicled fibula transfer . After aAfter the first flap coverage, the patient developed an infection at the plate site. This could be attributed to the contamination of the medullary cavity by the exulcerated tumor. Although antiseptic surgical methods were employed and antibiotics were administered postoperatively, the infection developed probably due to the biofilm around the plate by an anaerobic bacterium which was not covered by the antibiotic treatment. After mobilization, an incongruent weight loading appeared to develop with instability on the lateral side of the fibula, which led to repeated microfractures and delayed union. Considering the contamination of the operative site and the difficult consolidation, an Ilizarov external fixator might have been an appropriate alternative for osteosynthesis from the start, as in the study of Khira et al. . NeverthThe transplantation of the fibula to a weight-bearing position implies periosteal hypertrophy, to match the size of the tibia, which is about double in size. The progressive transfer of weight bearing from the tibial tuberosity to the foot using the fitted boot assisted the fibular hypertrophy so that after 2 years, the fibula reached almost the diameter of the tibia.2 without partial necrosis [2, although we harvested two perforator flaps with the pedicle, marginal distal necrosis developed. This could be attributed to the insufficient perfusion of the distal part of the flap and the infection which developed around the osteosynthesis material. The resulting defect after revision could be swiftly closed with a normal-sized contralateral ALT perforator flap, despite the still healing split-thickness donor area. Except for the functional and aesthetical advantages, fasciocutaneous flaps develop a reliable collateral blood supply. In our case, we could split the extended flap longitudinally a year after transplantation, without any impairment of the blood supply.ALT perforator flap is one of the most reliable workhorse flaps in reconstructive surgery. Harvested as extended, the ALT can cover extremely large soft tissue defects. Saint-Cyr et al. reported over 12 ALT extended flaps with a mean surface of 365 cmnecrosis . In our In this case, we showed that in selected cases, the boundaries of limb salvage can be pushed far beyond the current standards of treatment. Our case presented a unique combination of pedicled fibula transplantation and free extended ALT perforator flap to reconstruct an extensive defect after resection of a rare cSCC on top of NL.Additional file 1: Video 1. The extension of the foot was actively possible


Intraspinal choristoma is a relatively uncommon intervertebral canal tumor. Prior to our reports, only 2 cases of intraspinal choristoma had been reported. Because this disease is not common and looks like a mass of fatty tissue on the magnetic resonance imaging (MRI), intraspinal choristoma can be easily misdiagnosed as teratomas or lipomas (like the case of this article presenting) without a pathology report. So if a lumber intraspinal lesion is discovered in a clinical examination, intraspinal choristoma should be considered as a differential diagnosis. We present a case of intraspinal choristoma that is unlike any other reported case.A 35-year-old woman with left lower extremity hypoesthesia and burning-like pain in the lumbar region for 1 month visited the local hospital for plain lumbar spine MRI. The patient was diagnosed with a lumbar space-occupying lesion. A second plain lumbar spine MRI scan and a MRI scan with enhancement were performed in our hospital to confirm the presence of a congenital lipoma in the spinal canal. A postoperative biopsy of the lumbar spinal mass indicated that the mass was an intraspinal choristoma located in the spinal canal.Intraspinal choristoma.The lesion was surgically removed, and follow-up plain and enhanced MRI images of the patient’s lumbar spine were obtained.After surgery, the patient no longer experienced the burning pain in her lumbar region or the left lower extremity hypoesthesia when the patient was discharged. And there was no evidence of recurrence 2 years after the surgery.The MRI presentation of intraspinal choristoma is similar to intraspinal lipoma. Therefore, a pathological assessment is critical to provide an accurate diagnosis. While it can occur anywhere in the body, it is more frequently observed in the head and neck region, especially in the pituitary gland and oral cavity. Intraspinal choristomas are rare and, to the best of our knowledge, only 2 cases have been reported.,5 Thus, it is pretty rare for choristoma to occur in the spinal canal. The postoperative pathological assessment of an intravertebral occupying lesion in our patient showed a case of intraspinal choristoma. In addition, this patient also had a combination of spinal cord deformities such as spinal cord tethering syndrome which makes this case unique.Choristoma is a tumor-like structure composed of normal tissue located at an abnormal site.A 35-year-old woman was diagnosed with an intraspinal space-occupying lesion after a magnetic resonance imaging (MRI) scan of her lumbar spine was performed at a local hospital. The presenting complaint was hyperalgesia of the left lower extremity and burning pain in the lumbar region for 1 month. The patient was admitted to our hospital for further diagnosis and treatment. The preoperative physical examination revealed scoliosis, pain elicited with positive pressure on both sides of the fourth lumbar vertebra, and bilaterally diminished Achilles tendon and knee tendon reflexes. The straight-leg raising test was positive. The extension-hallucis test was positive, and the tension test for the femoral nerve was negative.MRI of the lumbar spine revealed that a near-circular short T1 and a long T2 signal shadow with clear borders were observed at the level of the inferior border of the fourth lumbar vertebral body. A low signal of Short Time Of Inversion Recovery demonstrated a rounded, non-enhanced area at the level of the lower edge of the fourth lumbar vertebral body. The area exhibited clear borders and was approximately 1.0 cm in diameter within the spinal canal  was 14. Preoperatively, the lesion was thought to be a lumbar intraspinal lipoma. The mass was surgically removed under general anesthesia. During the surgery, the tumor appeared yellow and exhibited a tough texture. The patient’s burning pain in her lumbar region and the left lower extremity hypoesthesia disappeared after surgery. One month after surgery, the physical examination revealed that pain was not elicited with positive pressure on both sides of the fourth lumbar vertebra. The straight-leg raising test was positive, and the extension-hallucis test was negative. Furthermore, the bilateral Achilles tendon and knee tendon reflexes had returned to normal. The JOA score was 27 at discharge, and the improvement rate was 86.7%. We conducted a 2-year follow-up examination, and the JOA score was 28 in the second year after surgery, and the patient’s improvement rate was 93.3%. An MRI scan was taken 2 years after the surgery, and there was no evidence of recurrence  should be considered choristomas.,22 Malela et al reported a sporadic case of “Congenital Midline Spinal Hamartoma in a 5-Month-Old Infant in 2020.” The similarity with our case is that it also contained numerous adipocytes, however, the case reported by Malela et al did not exhibit cartilage, skeletal muscle, mature collagen fibers, nerve and vascular components in the normal tissue at the location of the choristoma. The histologic examination of our case revealed mature fibrous tissue and squamous epithelium, as well as small sweat gland ducts. These 3 mature tissue components should not be present in the spinal canal, so the histologic examination indicated that a choristoma was present in the patient in this report.Based on the pathological diagnosis, the features of choristoma and hamartoma can be confounding. Both are rare, and many clinicians and pathologists find it challenging to identify them accurately. In addition, many articles have mentioned the need to be able to differentiate between choristoma and hamartoma. However, none of the previous reports have clarified the critical features that facilitate differentiation. The term choristoma is derived from the Greek root “choristos,” meaning separated and also means mass or tumor. The term is synonymous with heterotopia and ectopia, which are defined as the proliferation of normal cells in an abnormal location There are 4 histological teratoma variants: mature teratoma, immature teratoma, malignant teratoma, and monodermal teratoma.Since choristoma is extremely rare, it is not usually suspected before the surgery and pathology. Alternative differential diagnoses include teratoma and intraspinal lipoma. It was noted that the preoperative imaging presentation of this patient was similar to intraspinal lipoma. Teratomas contain cell populations from at least 2 of the 3 primary germ layers, endoderm, mesoderm, and ectoderm. They form during embryonic development and may be diagnosed at any age. Teratomas are thought to result from the failure of primitive germ cells to migrate and usually occur along the body midline, anywhere from the coccyx to the pineal gland. The primitive germ cells fail to degenerate and subsequently proliferate in abnormal locations in the midline to form tumors. Thus, the pluripotent capacity of these cells gives rise to multiple tissues consisting of immature cells that have the potential for malignant degeneration. Lesions often are located in the lumbosacral region, while occurrence within the dura of the cervical and thoracic medulla is less common. Though the pathogenesis of intradural lipomas remains unclear but occasional association with other midline anomalies points to a developmental origin. Intraspinal lipomas often occur in conjunction with a tethered spinal cord, and the lesion may contain other types of tissues such as bone, cartilage, or even bone marrow. Ammerman et al had described the clinicopathological characteristics of intramedullary lipomas: the tumors tend to be subpial in origin, involve multiple spinal cord segments as well as adjacent nerve roots, and have no clear plane demarcating themselves from adjacent neural parenchyma. Histologic investigation classically demonstrates normal adipose cells with intervening collagen. The collagen is often in continuity with adjacent coverings of vessels and neural structures. As no clear cleavage plane is found between the intraspinal lipoma and spinal cord, very few reports recommend complete surgical removal of an intraspinal lipoma as complete resection is associated with significant postoperative complications. Our literature review revealed that patients with intraspinal lipoma have masses that exhibit an irregular morphology and no clear boundary with the spinal cord on preoperative MRI scans. Whereas in this case, the tissue mass presented a more round shape and a more distinct boundary. These characteristics might help differentiate choristomas from intraspinal lipomas using MRI in the future. These observations also suggest that surgery for choristomas differ from lipomas in that choristomas can be less aggressive, be completely resected, and have a predictably lower postoperative recurrence rate than intraspinal lipomas. In contrast, intraspinal lipomas are more closely related to peripheral nerve tissue. It is possible to reduce the size of an intraspinal lipoma by reducing the amount of body fat, which would alleviate the symptoms of spinal cord or cauda equina irritation caused by the tumor. Thus, it is worth considering whether weight loss could be used to reduce the tumor size in patients diagnosed with a suspected choristoma who are unwilling or unable to undergo surgery.Intraspinal lipoma also is a rare benign tumor, accounting for <1% of all spinal cord tumors.Although microscopic findings can be highly suggestive of cartilaginous choristoma of the tongue, immunohistochemical staining is essential to rule out other possible diagnoses and confirm the hypothesis. For immunohistochemical staining, S-100, GCDFP-15, CK7, CK8, P63, and AR are used as positive indicators for choristoma. Positive GCDFP-15 staining suggested the presence of apocrine sweat glands. Positive staining for CK7 and CK8 suggested epithelial tissue origins, while positive staining for S-100 was indicative that the tumor originated from neurological tissue. P63 is a specific and sensitive marker for human myoepithelial cells, and positive staining suggests that the lesion is more likely to be a benign tumor. Therefore, the results that were obtained confirmed the diagnosis of choristoma. Besides, a case of autograft-derived spinal cord mass following olfactory mucosal cell transplantation in a spinal cord injury patient, enlightened us that the possible etiology of this intraspinal choristoma is a malformation during the embryonic development, and the malformation results in a transplantation of the regional disordered embryonic cells in lumbar region. Therefore, we believe that the abnormal developmental process of spinal bifida is most likely the reason for the development of intrasipnal choristoma in our case. Based on this case, if a space-occupying lesion in the spinal cord with a predominantly fatty signal appears on the MRI, surgeons should carefully distinguish whether the boundaries between the occupying lesion and the surrounding tissues are distinct. If the lesion has definitive boundaries and the patient has congenital spina bifida or spinal cord embolism, a possible diagnosis of intraspinal choristoma should be considered. Surgical options include complete resection, and the prognosis for patients with intraspinal choristoma is generally considered to have a lower recurrence rate than an intraspinal lipoma.We also suggest that it was not a coincidence that the patient also exhibited spina bifida and spinal cord tethering, as an association between spinal dysraphism and the occurrence of a lipoma is possible. The etiology of congenital spina bifida is not fully understood. However, most scholars believe that abnormal embryonic development of the neural tube is responsible for spina bifida, and abnormalities in the fusion process result in its formation. Specifically, the neural tube separates from the ectoderm through a process of detachment. After separation, midline fusion of the ectoderm above the neural tube occurs. Because choristoma is classified as a benign tumor, many studies do not discuss recurrence. The case reported in this study showed benign tumor characteristics based on the preoperative MRI findings and the intraoperative observations. We followed the patient in this case for 2 years, and the patient did not exhibit any recurrence, which indicated that surgical excision is curative, with a low incidence of recurrence. However, a case of osseous choristoma that exhibited recurrence was reported in another article, which might have been due to the tumor having a greater degree of malignancy. Typically, tumors that are multiple and have no pedunculated mass surrounded by soft tissue, such as within the buccal mucosa or muscle, might have the possibility of recurrence.In general, the clinical course and histopathological features of intraspinal choristoma indicate its benign nature. In 2 reported cases in children, the tumor was incompletely excised. One child was followed for 7.5 years and the other for approximately 16 months, without evidence of recurrence or residual tumor.Intraspinal choristoma is a rare congenital intraspinal tumor that might correlate significantly with congenital spinal deformities. Intraspinal choristomas are not easily distinguishable from intraspinal lipomas and intraspinal hamartomas based on MRI. However, if the lesion has distinct boundaries and the patient has congenital spina bifida or spinal cord embolism, a possible diagnosis of intraspinal choristoma should be considered. With the presence of distinct tumor boundaries, surgical options for complete resection should be considered. The prognosis of a lower recurrence rate for an intradural psammoma is generally better than for an intraspinal lipoma.The authors express their gratitude to all of the staff of the China-Japan Union Hospital of Jilin university who were involved in this patient’s care.Resources: Yongchuan Guo, Qianlei Liang,Yan Wang,Liang HanSupervision: Yongchuan Guo, Qianlei LiangWriting – original draft: Jinxin YangWriting – review & editing: Jinxin Yang


Low‐grade fibromyxoid sarcoma is a rare painless neoplasm that primarily grows in young adults' proximal extremities and trunks. The lungs are infrequent sites for this type of sarcoma.We reported a 26‐year‐old female that presented with a chief complaint of chest pain from a few months ago to Kasra hospital, Tehran, Iran, in August 2021. Chest computed tomography (CT) showed a hypodense mass with a well‐defined margin measuring 9.3 cm in the left upper lobe and multiple hypodense lesions with a lobulated appearance with a total diameter of 15.5 × 13.5 cm in the left lower lobe of the lung.This is the largest case of primary pulmonary low‐grade fibromyxoid sarcoma (30 × 28 × 7 cm), which seemed unresectable at first evaluation. Due to the extent of the tumor, left pneumonectomy was performed, leading to attenuation of symptoms and no recurrence at a six‐month follow‐up. She had also experienced progressive shortness of breath afterward. On the first admission, physical examination showed no abnormal findings, except for decreased auscultation of the left lung. Chest X‐ray revealed a huge circular, homogenous mass lesion occupying most of the left hemithorax Figure . Follow‐The pneumonectomy specimen consisted of a well‐circumscribed, homogeneous tumor measuring 30 × 28 × 7 cm with a portion of the lung measuring 10 × 8 × 4 cm and a small portion of the bronchus (1.5 × 1.2 cm) ; investigation ; project administration ; supervision ; validation ; visualization ; writing – original draft ; writing – review and editing . Matin Vahedi: Conceptualization ; investigation ; project administration ; supervision ; validation ; visualization ; writing – original draft ; writing – review and editing . Behnaz Jahanbin: Data curation ; investigation ; validation ; writing – review and editing . Fatemeh‐sadat Tabatabaei: Data curation ; investigation ; writing – review and editing . Shahab Rafieian: Conceptualization ; investigation ; project administration ; resources ; supervision ; validation ; visualization ; writing – review and editing .The authors have stated explicitly that there are no conflicts of interest in connection with this article.Investigations were in accordance with the Helsinki Declaration of 1964 and all subsequent revisions.Informed consent was obtained from the patient to publish case details and use images.


Radiological presentation of central nervous system (CNS) aspergillosis is variable and depends on the immune status of the patients. Typical features of meningoencephalitis, infarction, abscess, and mycotic aneurysms commonly occur in immunocompromised patients. A rare mass-like or tumoral form of cerebral aspergillosis has been described mostly in immunocompetent patients which results in a diagnostic dilemma, thus potentially causing a delay in the management. We present a case of a large CNS aspergilloma mimicking an infiltrative callosal neoplasm in a young immunocompetent patient. Careful evaluation of imaging features, anatomical location, enhancement pattern, concomitant sinonasal and orbital extension, and angio-aggressive nature of the mass lesion with a high index of suspicion can help diagnose CNS aspergillosis in such patients. Aspergillus species are the most common cause of central nervous system (CNS) fungal infections, with Aspergillus fumigatus being the most frequent cause of invasive aspergillosis in immunocompromised hosts and Aspergillus flavus, due to greater virulence, the most frequent cause in immunocompetent patients. Clinical manifestations of the disease process depend on the extension of the infection. Symptoms can range from headaches and seizures to focal neurological deficit [ deficit ,2.Radiological presentation of cerebral aspergillosis varies from that of meningoencephalitis, cerebral infarction, cerebral abscess, or mycotic aneurysms, which are findings typically seen in immunocompromised patients. A rare mass-like or tumoral form of CNS aspergillosis has also been described in the literature, mostly encountered in immunocompetent patients, which results in a diagnostic dilemma as the possibility of fungal infection is considered less likely in such patients which can cause a delay in management . We pres9/L .A 26-year-old male, with no known comorbidities, presented to the neurology clinic with complaints of headache and blurring of right-sided vision for the past two months. He had no medical or surgical history. He belonged to the lower socioeconomic stratum, lived on the outskirts of the city, and was a landlord by profession. He had no significant occupational or exposure history except for the presence of cotton factories near his residence. He had no sinonasal symptoms. He was vitally stable with unremarkable general and systemic examinations. His blood workup was normal; the complete blood count showed a total leukocyte count of 8.6 × 10Magnetic resonance imaging (MRI) performed with a 3-Tesla scanner showed a large infiltrative enhancing lesion in the anterior cranial fossa. It was infiltrating bilateral frontal lobes involving the genu of the corpus callosum and extending into the right-sided basal ganglia. There was an associated mass effect and surrounding edema. The lesion appeared heterogeneously iso to hypointense on T2-weighted sequences and isointense on T1-weighted sequences with avid post-contrast enhancement. There was subtle patchy peripheral diffusion restricted foci within it, with no significant susceptibility on susceptibility-weighted imaging (SWI)  scan showed residual fungal tissue, and he was treated with intravenous voriconazole. Since then, the patient has presented to the emergency room twice with complaints of progressively increasing seizures of up to four to six episodes per day as of last year and is currently being managed conservatively on lacosamide and levetiracetam. His follow-up imaging after a year of treatment showed significant improvement in the disease process with associated encephalomalacia and gliosis at the site of surgical resection Figure .The radiological presentation of CNS aspergillosis is variable and depends upon the immune status of the patient. The characteristic findings described in immunodeficient patients can range from ring-enhancing lesions, abscesses, meningitis/meningoencephalitis, and small ischemic or hemorrhagic infarcts due to vasculitis and mycotic aneurysms . In immuThe diagnostic hint for CNS aspergillosis in our case was the hypointensity in the lesion on T2-weighted sequences on MRI and infiltration into the ethmoid sinuses suggestive of an aggressive process with paranasal sinus involvement. The T2 hypointensity correlates with the presence of paramagnetic elements such as iron, manganese, magnesium, and zinc that are essential for the hyphal growth of the fungi . StrikinWith regards to the differentials of neoplasms of the anterior cranial fossa, lymphoma and meningioma have been described as mimickers of CNS aspergillosis ,8,9. VerCNS lymphoma can also involve the paranasal sinuses with coexisting intracranial components, which may be difficult to differentiate from aggressive fungal infection. Lymphomatous lesions usually appear hyperdense on CT and may also be hypointense on T2-weighted sequences on MRI due to high cellular pattern. They exhibit homogenous and avid post-contrast enhancement similar to aspergilloma; however, the involvement of paranasal sinuses, encasement and involvement of the internal carotid arteries, and adjacent calvarial involvement are not frequently encountered. MR spectroscopy demonstrates elevated lipid/lactate peaks and high choline/creatine ratios, and diffusion-weighted imaging shows restriction, which can be differentiating features from CNS aspergilloma . In our In contrast to aspergilloma, meningioma appears isointense to gray matter on both T1- and T2-weighted sequences. Both lesions enhance homogeneously on post-contrast sequences. Rarely, meningioma can also arise in the orbit and paranasal sinuses . FibrousIn the past, CNS aspergillosis was generally considered a fatal disease. Medical advances have reduced the mortality rate but early diagnosis is crucial. Culture and histopathology remain the gold standard of diagnosis by obtaining infected tissue. Molecular methods and biochemical laboratory analysis such as 1,3)-beta-d-glucan and galactomannan levels can also be used for diagnosis . Treatme,3-beta-dFungal infections such as cerebral aspergillosis can occur in immunocompetent individuals with no prior history and may present as an intra-axial mass-like lesion and involve the corpus callosum, therefore mimicking a cerebral neoplasm. This form of presentation usually poses a diagnostic dilemma as the possibility of fungal infection is considered less likely in such patients causing a delay in the management. Careful evaluation of MRI features such as signal intensity on different sequences, anatomical location, enhancement pattern, concomitant sinonasal and orbital extension, and angio-aggressive nature of the mass can help diagnose CNS aspergillosis in such patients.