E. coli on culture. The patient underwent left nephrectomy, and the histopathological report of the surgical specimen confirmed XGP.Xanthogranulomatous pyelonephritis (XGP) is a rare variant of chronic pyelonephritis. It is characterized by progressive parenchymal destruction caused by chronic renal obstruction due to calculus, stricture, or rarely tumor, resulting in kidney function loss. Herein, we describe the case of a 36-year-old female who presented with left loin pain, left lower limb pain, and dysuria. On contrast-enhanced computed tomography (CECT), multiple abscesses and an obstructive staghorn calculus were depicted in the left kidney with the classical appearance of “Bear Paw Sign.” An abscess with calculi was also present within the left psoas muscle. Though psoas muscle abscess in association with XGP was described, a ureteric fistula and calculi within the psoas muscle have not yet been reported in the literature. Left nephrostomy was performed, which came out to be positive for  It is more frequently seen in middle-age females, and elderly.3/µI  and anemia - Hemoglobin of 8.2 g/dL . Renal Function test and serum electrolytes were within normal limits. The urinalysis revealed 10-15 leukocytes/HPF . On a plain radiograph, there was a large radio-opaque shadow in the left renal fossa  showed a large calculus, probably a staghorn calculus measuring ~ 3.0 cm with a twinkling artifact and posterior acoustic shadowing 2B. Also,The CECT depicted an enlarged left kidney with gross hydroureteronephrosis with thinning of the renal cortex and a staghorn calculus measuring ~ 3.8 × 2.4 cm within the renal pelvis. The renal pelvis appeared contracted, whereas calyces were dilated, giving the appearance of a “Paw of a Bear,” referred to as “Bear Paw Sign” A. Also, Escherichia coli (E. coli) was isolated on culture. After 5 days, a left-sided nephrectomy was performed, and the specimen was sent for histopathology examination. The surgical specimen weighed around 150 g . The excised kidney measured 10.0 × 7.0 × 4.0 cm and was partially covered with perinephric fat. The attached ureter measured ~ 4.0 cm in length. The cut section of the kidney showed a staghorn calculus filling the pelvicalyceal system measuring 3.5 × 1.0 cm. Also, a few areas of yellowish discoloration were noted within the pelvicalyceal system, probably due to xanthoma cells (The working diagnosis based on the imaging exams was left XGP with psoas abscess. Left nephrostomy was performed, and purulent fluid was drained, from which ma cells 44C.Microscopically, the kidney sections showed hyalinized glomeruli and atrophic tubules lined by flattened cells, and the surrounding blood vessels were thickly walled. Interstitial fibrosis with a dense collection of inflammatory cells composed of sheets of lymphocytes, plasma cells, neutrophils, histiocytes, and multinucleated giant cells were seen. Islands of foamy macrophages, foci of calcification, lymphoid follicles, and thickened capsule were also seen. At some places, the inflammatory cells and the proliferating capillaries were overlined by a transitional epithelium with ulceration. AD.Proteus mirabilis, E coli, or Staphylococcus aureus. The annual incidence of XGP is 1.4 per 100,000. XGP is a rare variant of pyelonephritis and is seen more commonly among females.,-XGP is a suppurative granulomatous infection characterized by progressive parenchymal destruction caused by chronic renal obstruction due to calculus or stricture or rarely tumor, resulting in a non-functioning kidney. XGP accounts for 0.6% to 1% of all pyelonephritis cases, globally. The common organisms on urine culture are ,,E coli, which is the most frequent bacterial cause of urinary tract infection.CT is the gold standard diagnostic method for XGP because it demonstrates the specific intrarenal findings and shows the extrarenal extension, which is useful for surgical scheduling.The patient was prescribed broad-spectrum antibiotics for 5 days, followed by total left nephrectomy. Usually, on MRI, XGP appears as a hyper signal on T1- weighted image due to the lipid-laden foamy macrophages and shows a signal void if calculi are present within the collecting system. However, this MRI sign is not always reliable if the lesion lacks a certain amount of xanthoma cells.,,Medical and surgical management plays an important role in the treatment of XGP. In cases of unilateral diffuse XGP, nephrectomy is the usual therapeutic option. While in cases of bilateral XGP, when surgery is not feasible or in patients with focal XGP, the broad-spectrum antibiotic regimen is indicated.XGP is an uncommon entity associated with urinary tract obstruction and infection. Also, its association with calculus migration into psoas abscess is an unusual complication. It is difficult to differentiate it from renal cell carcinoma on imaging, and hence histopathological correlation is required. Imaging features are not enough to confirm the diagnosis of XGP, but in this case, demonstration of “Bear paw sign” helped in ruling out other uncommon differentials. In this case, the correlation of the clinical findings with radiologic features, followed by proper treatment and histopathological correlation not only helped in confirming the disease but also improved patient recovery.


Anterior clinoidectomy (AC) is a key microsurgical step for the safe and successful management of parasellar pathologies that involve the anterior clinoid process (ACP) and the optic canal. Traditionally, extra and intradural ACs are performed separately according to the surgeon's experience or preference. The objective is to present and discuss the tailored AC concept through illustrative cases. We conducted a retrospective record review of three patients who underwent AC as a surgical step for the treatment of parasellar pathologies that involve the ACP and optic canal. A review of the relevant literature on AC was performed in the PubMed, LILACS, and SciELO databases.In all three cases, the pterional craniotomy was the preferred approach for AC. Case 1, a 47-year-old female patient with type III anterior clinoidal meningioma, underwent a tailored intradural technique  with total tumor resection and complete visual recovery. Case 2, a 63-year-old female patient with a complex type II anterior clinoidal meningioma with extensive hyperostosis of the ACP, underwent a hybrid AC technique with complete removal of the tumor and visual improvement. Case 3, a 62-year-old female, underwent a tailored intradural AC for clipping an incidental carotid-ophthalmic aneurysm.Tailored AC aims to provide adequate exposure with less risk of neurovascular injury, allowing enough space to safely treat parasellar lesions. The type, size, and location of the lesion, as well as the surgeon’s experience, should always be considered for surgical planning. The anterior clinoid process (ACP) is a triangular-shaped process of the sphenoid bone, which is a continuation of the lesser sphenoid wing and is medially connected to the sphenoid body by the optic strut and roof of the optic canal . ACP relAnterior clinoidectomy (AC) is a key microsurgical step for the safe and successful management of paraclinoid aneurysms and parasellar neoplastic lesions, increasing the surgical path through the opticocarotid and carotid-oculomotor triangles and allowing the decompression and safe mobilization of the optic nerve -5. Two mThe aim of this study is to present and discuss the tailored AC concept through illustrative cases.This study is a retrospective record review of three patients who underwent AC for the treatment of parasellar pathologies that involved the ACP and optic canal. Clinical and surgical records, ophthalmological examinations, which included computerized campimetry, radiological data as magnetic resonance imaging (MRI), computed tomography (CT) scan, and CT angiography, AC technique applied, and outcome were reviewed. A written informed consent form was obtained from all patients. A review of the relevant literature on AC was performed in the PubMed, LILACS, and SciELO databases.Case 01PresentationA 47-year-old female patient presented with gradual visual loss in the right eye over the last four months. Computerized campimetry revealed nasal hemianopia and superior temporal quadrantanopia in the right eye. Contrast-enhanced MRI showed a type III anterior clinoidal meningioma involving the optic nerve and extending into the optic canal Figure , panel ADescription of TechniqueThe patient was positioned in dorsal decubitus. The head was fixed in a three-pin device, elevated, turned 30 degrees to the contralateral side, and slightly extended. A pterional craniotomy was performed and the dura mater was opened. The Sylvian fissure was dissected and the frontal lobe gently retracted. The tumor was identified involving and compressing the right optic nerve Figure , panel COutcomeThe patient had a good outcome with visual improvement during the follow-up. Contrast-enhanced MRI showed total resection Figure , panel ECase 02PresentationA 63-year-old female patient presented with progressive visual loss in the left eye and facial hypoesthesia on the same side over the last seven months due to recurrent anterior clinoidal meningioma. The preoperative MRI in 2012, before the first surgery, revealed an extra-axial lesion occupying the opticocarotid cistern with homogeneous contrast enhancement and dural attachment in the superolateral aspect of the ACP suggestive of type II anterior clinoidal meningioma, with minimal hyperostosis of the ACP on bone window CT scan. During the first surgery, the soft portion of the tumor was removed, but not the bone infiltration. Histopathological examination confirmed meningioma grade I . Six years later, the MRI and CT scan showed intradural tumor recurrence with encasement of the ICA and extensive hyperostosis of the sphenoid wing and ACP (Figure Description of TechniqueThe patient was positioned in dorsal decubitus with an ipsilateral shoulder roll. The head was fixed in a three-pin device, elevated, turned 30 degrees to the contralateral side, and slightly extended. A pterional approach was performed. The sphenoid ridge was drilled and the meningo-orbital band was carefully cut to expose the ACP. Several anatomic bone changes were observed. Extensive extradural drilling of the lesser and greater sphenoid wing with a high-speed drill was performed Figure , panel COutcomeThe patient had a good outcome with visual improvement during the two-year follow-up. Contrast-enhanced MRI showed no residual tumor Figure , panel GCase 03Case PresentationA 62-year-old female patient presenting with recurrent scintillating scotoma in the right eye about 12 months ago. CT angiography revealed a 7-mm saccular paraclinoid aneurysm of the right ICA, with the neck measuring 3.5 mm (Figure Description of TechniqueThe patient was positioned in dorsal decubitus with an ipsilateral shoulder roll. The head was fixed in a three-pin device, elevated, turned 30 degrees to the contralateral side, and slightly extended. The standard pterional approach and durotomy were performed. The Sylvian fissure was opened and the optic nerve, ICA, and ACP were exposed. Until this step, a microscopic inspection was not able to view the aneurysm neck (Figure OutcomeThe patient's visual function was kept intact and postoperative CT angiography revealed complete aneurysm occlusion Figure , panel EThe AC was registered for the first time over 70 years ago using the intradural AC technique . In 1985The most frequent approach for AC reported in the literature is the pterional approach . HoweverAC is a key microsurgical step for the safe and successful management of paraclinoid aneurysms and parasellar neoplastic lesions, increasing the surgical path through the opticocarotid and carotid-oculomotor triangles and allowing decompression and safe mobilization of the optic nerve -5. Two mIntradural and extradural techniques are widely accepted, each one with relative advantages and disadvantages . The maiThe main advantage of the extradural technique is that it is entirely performed in the extradural space, allowing a layer of the dura to protect the adjacent eloquent structures and to avoid subarachnoid spreading of bone dust and postoperative headache . AdditioACP is generally composed of compact bone and is involved by clinoidal ligaments and dural elements, however, pneumatized ACPs are observed in up to 28% of the patients ,7. IntenAn intradural AC is indicated for small and soft intradural lesions around the ACP, such as type III anterior clinoidal meningioma or carotid-ophthalmic aneurysms, or when optic nerve mobilization is mandatory for greater exposure of the opticocarotid and carotid-oculomotor triangles to access lesions that extend to the upper clivus and interpeduncular fossa ,7,11-12.Extradural AC is commonly preferred for complex lesions of the lesser sphenoid wing and cranial nerve compression that cross through the superior orbital fissure and is especially used in large parasellar meningiomas with extensive bone involvement, which promotes early tumor devascularization . HoweverBeyond the scope of the intradural and extradural techniques, the concept of tailored AC has been developed to balance the risks of total removal and to avoid more extensive and time-consuming techniques. It has been noted that ACP does not need total removal in all cases, and custom-tailored AC can be practiced to avoid potential mechanical and thermal complications ,13,20,30Lastly, skull base surgery has been modified in an attempt to make it less morbid -29. TailThrough three illustrative cases, we demonstrated that the "tailored AC " aims to provide adequate exposure with less risk of neurovascular injury, allowing enough space to safely treat parasellar lesions. The type, size, and location of the lesion, as well as the surgeon’s experience, should always be considered for surgical planning.


Dentigerous cysts (DC) are the most common odontogenic cystic lesions of inflammatory origin occurring in children. These lesions can be treated by enucleation with or without related impacted teeth or marsupialization/decompression. The latter procedures have been used successfully for many years, but decompression is increasingly recommended in children because of its good outcomes and the preservation of the developing tooth. This conservative surgical technic allows simultaneously the normal eruption of the involved permanent teeth and the ossification of the bony defect. The present report describes an 8-year-old female patient with an inflammatory DC associated with an endodontically treated primary molar accidentally discovered on panoramic radiograph. Decompression of the cyst was performed, by means of a removable space maintainer acting as an acrylic obturator after removal of teeth 83 and 84. Ten months after the decompression procedure, a complete resolution and regression of the cystic lesion with full spontaneous eruption of the premolar were observed. From the age of 5 to 12 years, children are in mixed dentition. At this age, to ensure proper eruption of the permanent dentition and to prevent arch length deficit and loss of space, it is important to preserve the primary teeth [Several disturbances can cause either infection or necrosis of the pulp tissue of primary teeth, and since pulp therapy is not always easy to perform on these teeth because of their complex anatomy, the preservation of primary teeth can induce undesirable effects on the permanent successor, such as the formation of inflammatory DC .These cysts are caused by the accumulation of fluid between the reduced enamel epithelium and the crown of the tooth . AlthougEnucleation with or without impacted teeth or marsupialization/decompression are the treatments of choice of inflammatory DC. Both procedures, marsupialization and decompression, have been used successfully for many years, but decompression is increasingly recommended because of its successful outcomes and preservation of the developing tooth structure .Since these cysts are more common in children, conservative management by decompression may be recommended, allowing the conservation of the permanent involved tooth , 8.The present report is aimed at describing a complete regression of an inflammatory DC with full spontaneous eruption of the premolar ten months after a decompression procedure in an 8-year-old female patient.The present clinical case report was reported according to the CARE (CAse REport) Guidelines .An eight-year-old female patient consulted the Department of Pediatric and Preventive Dentistry at the Faculty of Dental Medicine of Monastir for dental malocclusion management.Written informed consent was obtained from the child's parents for all imaging exams, treatment modalities, and data publications.The panoramic radiograph performed for the interceptive orthodontic treatment revealed a large unilocular radiolucency repressing the dental germ of the first lower-right premolar (#44) including the lower-right deciduous canine (#83), and associated with a treated root of nonvital lower-right first primary molar (#84) .The medical history of the patient did not reveal any specific systematic diseases or previous traumatic injuries in the affected area.The extraoral examination seemed to show no abnormalities while the intraoral examination revealed an imperceptible expansion of the buccal cortical on the alveolar ridge in relation to teeth 83 and 84.Inspection showed a normal looking mucosa . PalpatiA retroalveolar radiograph was performed and showed a well-defined surrounded by a slight peripherical radiopaque thickening lesion apical to the lower-right first primary molar (#84) .To confirm the lesion limits, a cone-beam computed tomography (CBCT) was requested.The sagittal and coronal views of the CBCT revealed a 12 mm diameter well-demarcated unilocular radiolucency Figures  and 5.The lesion was identified in the mandibular right region causing expansion of the buccal cortical with no signs of root resorption in the adjacent tooth and surrounding the crown of the unerupted first premolar (tooth 44).The first lower-right premolar germ was forced against the lingual cortical with a mesial angulation. Based on the above-mentioned clinical and radiological findings, a provisional diagnosis of inflammatory DC was made.The second primary molar and canine were extracted under local anesthesia, and a conservative approach by decompression to preserve the mandibular first premolar was followed.The extraction socket was extended to establish a communication between the cyst and the oral cavities without disturbing the erupting premolar .The cystic fluid was evacuated under irrigation with normal saline solution through the socket.An iodoform gauze was then packet into the lesion cavity, and sutures were placed.The patient and her parents were advised to follow postsurgical instructions, rinsing the opening cyst twice a day with saline solution.For the first 48 hours, postoperative care included the use of a cold pack. For pain management, 200 mg dexibuprofen three times a day was prescribed for the young patient.Two days postoperatively, an impression was made before gently removing the gauze which helped to prevent the alginate from getting the bone cavity. Then, a removable space maintainer acting as an acrylic obturator was designed .During the surgical procedure, a tissue sample was taken for biopsy. Microscopically, the cyst was lined with cuboidal nonkeratinized stratified epithelium resembling reduced enamel epithelium, and the underlying connective tissue capsule showed chronic inflammatory cell infiltration. The histopathological examination confirmed the initial diagnosis of the inflammatory DC.The acrylic obturator remained until the eruption of tooth 44 was visualized using a subsequent radiographic evaluation.The follow-up appointments were scheduled every three months postsurgery.The three-month postoperative radiograph showed a reduction in radiolucency associated with a gradual spontaneous eruption of the tooth .After 10 months follow-up, the radiographic and clinical findings showed that the affected teeth successfully erupted without any intervention.The tooth was vital with almost a complete root formation Figures  and 10.According to the World Health Organization (WHO) classification of odontogenic lesions, odontogenic cysts were divided into developmental and inflammatory cysts. The inflammatory odontogenic cysts included radicular cysts and inflammatory collateral cysts while the developmental odontogenic and nonodontogenic cysts included dentigerous cysts, lateral periodontal cysts, botryoid odontogenic cysts, gingival cysts, glandular odontogenic cysts, orthokeratinized odontogenic cysts, and nasopalatine duct cysts .The DCs constitute the most frequent maxillary cystic lesions (30% of the odontogenic cysts) , with soThese cysts are also very common in mixed dentition and are usually associated with the roots of a nonvital or necrotic primary tooth and the crown of an unerupted permanent tooth , such deThe pathogenesis of DC is still controversial. However, three possible mechanisms were proposed in the literature .The first mechanism suggested that the developmental DC might form a dental follicle and might become inflamed later . The secThe follicle of permanent successor might get secondarily infected from either periapical inflammation of a nonvital primary tooth or other source leading to a DC formation, and this inflammatory process may explain the DC development in the present report .For this reason, especially in mixed dentition, it is very important to establish not only a clinical but also a radiological follow-up of the endodontically treated primary molars until the eruption of permanent successors to be able to detect as soon as possible the side effects of pulp therapy and to manage them in prompt time.Usually, DCs are asymptomatic and are discovered during routine dental radiographies .Radiographically, these developmental cysts are characterized by the presence of a radiolucent, well-defined, unilocular lesion around the crown of an unerupted permanent tooth. They can be reported in the presence of exformation of the maxillaries, dental structures shifting, or delays in dental eruptions . In the According to the literature, the DC management is exclusively surgical, and two treatment techniques have been suggested: the enucleation or the marsupialization/decompression.In their retrospective study, Iatrou et al. choose the technique of enucleation, reserving the marsupialization to large cysts. An association of both techniques can be suggested for the more aggressive cysts to reduce the volume of the lesion, before surgical removal .Conservative techniques for the treatment of odontogenic cysts have evolved gradually the last decades.Marsupialization and decompression are minimal surgical intervention but decompression is less invasive because it requires a smaller bony window.Decompression may envelop marsupialization, and it includes any method that keeps an opening into the exterior by using a conduit to decrease intracystic pressure .The benefits of decompression are maintenance of pulp vitality, prevention of maxillary fractures, preservation of the inferior alveolar nerve or maxillary sinus, and low risk of recurrence .In the present reported case, since the patient and her parents were cooperative, the decompression was recommended.After decompression, the cystic cavity must remain open; numerous means have been described such as stents, stents fixed by mini-screws, packs of iodoform gauze, tubes for decompression, and retainers .Ghandour et al. recommended the placement of a gauze during the first two days after the surgery to keep the surgical site open . PreservIn mixed dentition, a loss of space may occur during the first three weeks following the extraction of the primary teeth; for this reason, maintaining the space is necessary to avoid the decrease in the arch length . In the A clinical and radiographic follow-up will be necessary, every three months initially, then every six months, until the end of the eruption of the tooth and the healing of the bone .Both radiological and clinical findings after 10 months showed in the present report that the first premolar had successfully erupted without any orthodontic intervention.Although the predictive criteria for this eruption remain unknown, the size of the cystic lesion does not constitute a risk factor for the eruption.In fact, the most favorable predictive factors are essentially the age of the patient (less than ten years old), the depth of the inclusion (less is better), and the angulation of the germ (inferior to 25°) .After minimal surgical intervention, the spontaneous eruption may occur in approximately 70% to 90 of cases. The length of the eruption is very variable, ranging from seven months to five years, and orthodontic traction can be associated , 21.Extraction of the primary infected tooth and decompression is the treatment of choice for infected dentigerous cyst in mixed dentition.The surgical procedure can be followed by wearing of an acrylic resin obturator, incorporating features of space maintainer especially when primary molars are involved, both to maintain the surgical opening during healing and to prevent the arch length deficiency.This report highlights the interest of performing a systematic radiological examination during the child's first visit.


Primary testicular lymphoma is a rare but aggressive form of extranodal lymphoma.A 48-year-old man came with painless lump on both testicles since 10 months ago. Testicular tumour marker revealed increased LDH. Testicular USG revealed semi-solid spongiform mass in the right epididymis and bilateral testicles, suggesting malignancy and minimal bilateral hydroceles. Histopathologic examination revealed diffuse large B-cell type lymphoma.Orchiectomy alone is not the definite treatment. Chemotherapy was given to increase survival rate. It is important to diagnose the disease with adequate diagnostic work up to achieve better prognosis and early treatment. Primary testicular lymphoma (PTL) is a rare but aggressive form of extranodal lymphoma. It accounts for 1–2% of all non-Hodgkin’s lymphomas and 4% of extranodal lymphomas. Most PTL is diffuse large B-cell (DLBLC) type, which has the potential for aggressive clinical behaviour [A 48-year-old man presented with a lump on both testicles since 10 months prior to admission. The patient neglected it and did not seek any treatment due to lack of pain and the patient thought that the size of the lump was tolerable. The lump grew bigger in the last 3 months.The patient smoked half a pack of cigarettes per day. Previous history of hypertension, diabetes, heart disease and drug consumption. No familial history of malignancies was found.Vital signs were within normal limits. Scrotal examination revealed enlarged hard masses affecting both testes and right epididymis, with right mass was larger than the left.Complete blood count of the patient was unremarkable. Testicular tumour markers revealed increased in LDH (314 U/L), normal AFP  and normal bHCG . Chest x-ray showed normal result with no lung metastasis. Ultrasonography (USG) examination revealed semi-solid spongiform mass in the right epididymis and bilateral testicles, suggestive malignancy and minimal bilateral hydroceles . BilaterDuring surgery, a brownish-white tumour mass was found filling the entire testicular tissue. On the right testicle, there was adhesion of the mass to the surrounding tissue, but it can still be released. Only right spermatic testicular cord appeared infiltrated with mass.Macroscopic examination of the excised tumours showed enlarged right testicle with lamellar solid white mass attached to spermatic cord cyst; enlarged left testicle weighed with lobulated mass on lamella . HistopaTesticular tumour marker and abdominal pelvic CT scan were performed during follow-up after the surgery to detect any remainder of the malignancy. Compared to pre-surgical result, the testicular tumour marker showed a normal LDH (222 U/L), normal AFP  and normal bHCG . Testicular tumour marker was obtained before histopathological result was available. CT scan result showed non-homogenous lesion with clear borders on the right scrotum , no visible residual mass in the left testicle and multiple lymph node enlargement with partial conglomeration in the paraaortic region.The working diagnosis was bilateral testicular tumour (right testicle grade pT3N2M0S1 and left testicle tumour grade pT2N2M0S1). Right tumour invaded the spermatic cord, and the left was limited to the testis and epididymis. Both tumours invaded surrounding lympho-vascular system. No metastasis was found.International Prognostic Index (IPI) score of 2. Written informed consent was obtained from the patient for this case report.Poly-chemotherapeutic treatment was initiated: six cycles of Rituximab, Cyclophosphamide, Doxorubicin and Vincristine. There was no mass nor metastasis found after the treatment, confirmed by whole abdominal USG in June 2020. The patient was included in the intermediate risk group  with PTL usually presents as unilateral , firm anDiagnosis is based on history taking, physical examination and USG imaging. HIV serology should better be performed as it has high concurrency with Non-Hodgkin Lymphoma . In thisBlood–testis barrier makes testicular tumours inaccessible to systemic chemotherapy, but it can be removed by orchiectomy. However, even in stage I disease, orchiectomy alone is not a definite treatment as in testicular DLBCL the risk of contralateral testicular relapse is high, 42% at 15 years among patients who did not receive prophylactic radiation in the IELSG study , 9. PropThe patient was given Rituximab, Cyclophosphamide, Doxorubicin and Vincristine as chemotherapy agents. Beside Rituximab, other common combination are cyclophosphamide, vincristine, doxorubicin and dexamethasone . MethotrStudies reported that PTL is a rare malignancy with poor outcome . Ann-ArbIn conclusion, it is important to diagnose DLBCL with adequate history taking, physical examination, and imaging modalities to achieve better prognosis with early treatment. Family physician or in primary care should also educate the patient about the early symptoms of tumour.


Idiopathic spontaneous pneumoperitoneum is caused by free air in the peritoneum when no established cause has been diagnosed. We present the case of a 61-year-old male with idiopathic spontaneous pneumoperitoneum, which started as abrupt abdominal pain. He described burning abdominal pain radiating to his right shoulder and endorsed symptoms of nausea, abdominal bloating, and heartburn but denied fever, chills, or vomiting. Chest radiograph and computed tomography demonstrated massive amounts of free air under the diaphragm, concluding an extensive pneumoperitoneum. He was diagnosed by standard imaging modalities and then underwent diagnostic laparoscopy, which did not reveal any areas of perforation. Subsequently, the patient had an uncomplicated recovery. The complexity of diagnosis and treatment has made it difficult for surgeons to treat spontaneous pneumoperitoneum patients.  Pneumoperitoneum is defined as free air within the peritoneal cavity with a majority of causes resulting from intra-abdominal viscus perforation . Peptic When there is no evidence of visceral perforation and indication for surgery, the diagnosis of exclusion is called spontaneous pneumoperitoneum (SP) or "non-surgical" pneumoperitoneum. The causes of SP are relatively complex but can involve intrathoracic, intra-abdominal, gynecologic, or iatrogenic causes . The diaA 61-year-old male patient with a past surgical history of appendectomy, cholecystectomy, and Nissen fundoplication  presented to urgent care with complaints of abdominal pain that started abruptly. He described burning abdominal pain radiating to his right shoulder and endorsed symptoms of nausea, abdominal bloating, and heartburn but denied vomiting. The patient confirmed flatus and bowel movement since the time of onset. He reported having a recent left rotator cuff repair, without complication, and denied taking nonsteroidal anti-inflammatory medications in his postoperative period. He was given a treatment of “GI cocktail” with 30 mg of toradol intramuscularly. Subsequent X-ray was obtained, which demonstrated an extensive pneumoperitoneum. Also, diffuse mildly conspicuous gas-filled loops of large and small bowel were noted throughout the abdomen with no convincing portal venous gas (Figure In the emergency department, the patient appeared to be in progressive distress and was found to have significant abdominal tenderness in all four quadrants, worse in the epigastrium. His focused abdominal examination was significant for guarding, distention, and tympany to percussion. Subsequently, general surgery was consulted for further management. CT was ordered Figure . FurtherThe laparoscopy showed no evidence of contamination, peritonitis, or perforation throughout the abdomen and the remaining gas was suctioned from the abdomen before closing. The patient’s symptoms resolved over the course of a couple of days in postoperative recovery. X-ray and CT imaging were repeated prior to discharge and demonstrated significant improvement of the pneumoperitoneum, with only a small volume of gas remaining Figures , 4. An ePneumoperitoneum with perforated intra-abdominal viscus accounts for 90% of all causes, with the remaining cases labeled as idiopathic SP. Risk factors for perforations include diverticulosis, obstruction, advanced age, and existing weaknesses in the colonic wall that have resulted from inflammatory bowel disease, infection, or malignancy -4. PatieThe vast majority of pneumoperitoneums are caused by retained postoperative air, but other sources of free air include mechanical ventilation, cardiopulmonary resuscitation, and pneumothorax ,6. ExcesThe diagnosis of SP requires a detailed history and physical examination, with further imaging including chest radiograph and CT. Plain chest or abdominal radiograph as well as CT will show free subdiaphragmatic air. If the patient has a perforation, CT findings predict the site of gastrointestinal tract perforation with 86% accuracy . Also, oConservative management should be the mainstay for patients with SP with minimal symptomology, with some studies suggesting even so when signs and symptoms of peritonitis are present ,15. No cWe presented a case of idiopathic SP in a 61-year-old male who presented with abrupt abdominal pain. He underwent diagnostic laparoscopy after multiple imaging modalities, but no evidence of visceral perforation was noted. The complexity of diagnosis and treatment has made it difficult for surgeons to treat SP patients. Of note, patients can undergo laparoscopic exploration as an alternative to laparotomy to minimize postoperative complications. Our patient reported an uncomplicated recovery and denied any recurrence of symptoms after his diagnostic laparoscopy.


Pulmonary hamartomas are benign lung tumors. They are uncommon and represent a small percentage of all solitary lung lesions. Hamartomas are composed of an abnormal mixture of epithelial and mesenchymal elements. They are usually discovered incidentally as patients are asymptomatic in most cases. On the other hand, sclerosing pneumocytomas are rarely discovered and are one of the most uncommon benign lung tumors. Herein, we present the case of a 37-year-old female who presented with hemoptysis. A computed tomography scan of her thorax revealed a right-sided round lesion in the interlobar fissure with no additional findings. Surgical excision was performed, which demonstrated an uncommon and unique finding of a pulmonary hamartoma coexisting with a sclerosing pneumocytoma found on exploration. This highlights the importance of exploration during surgical procedures in order identify missed lesions on imaging. Pulmonary hamartoma (PH), also known as mesenchymoma, was first described by Albrecht in 1904 . PHs usuOur patient is a 37-year-old female who presented with a three-day history of dry cough associated with hemoptysis. The hemoptysis started a day before presentation to the hospital and it occurred twice. The patient quantified the amount equal to a teaspoon full on each instance. She was otherwise asymptomatic with no associated fever, night sweats, or weight loss. Physical examination was completely unremarkable with clear lung fields on auscultation. Laboratory investigations, including a full blood count, renal function, electrolytes, and inflammatory markers, were also within normal limits. Her past medical history was unremarkable and she never smoked cigarettes. There was no additional history of pets at home or occupational exposures. Plain chest radiography revealed a right middle zone elliptical opacity with a small round shadow within Figure . She wasA CT scan was performed which demonstrated a loculated effusion in the upper part of the oblique fissure with a well-defined, rounded, soft tissue density lesion embedded within and measuring approximately 7 x 5 cm without calcifications Figure . This case was later discussed at a multidisciplinary team meeting followed by a plan to proceed with exploration and excision of the lesion.Surgery was initially performed by the means of a thoracoscopy through a 12 mm port. The interlobar fissure cyst containing the lesion was large in size (10 x 7 cm); therefore, it proved to be difficult to remove while keeping the cyst intact by thoracoscopy. This was important in order to avoid a sac rupture with the remote possibility of it being a hydatid cyst. Eventually, a right posterolateral thoracotomy through the fifth intercostal space was performed and the cyst dissected with complete excision Figure .Surprisingly, during exploration, another mass was noted in the upper lobe ipsilaterally and was excised. The wound was then closed and both lesions were sent to the laboratory for histopathological examination. There were no immediate complications and the recovery period postoperatively was unremarkable, alongside a complete resolution of the hemoptysis and cough.The first soft tissue lesion resected from the interlobar fissure measured 2 x 1.5 x 1 cm. Its histological findings were consistent with a pulmonary hamartoma Figure . The second lesion that was found on exploration in the upper lobe ipsilaterally measured 1 x 1 x 0.3 cm. On microscopic examination, it showed an ill-defined area of multiple thin-walled blood vessels with mild focal smooth muscle proliferation. The vessels showed positive immunohistochemical staining for the vascular endothelial marker. This was consistent with the diagnosis of sclerosing pneumocytoma, the angiomatous type Figure .Pulmonary hamartomas consist of a disorganized array of various connective and epithelial tissue normally found in the lungs. In a study by Cosio et al., it was concluded that they are the most common form of benign lung tumors, are male predominant, and peak in incidence in the fifth or sixth decade of life . They arThe mesenchymal components of PH are highly variable in histology and are composed of a mixture of mature mesenchymal tissue . They arPSP is a rare finding accounting for only 1% of all benign lung tumors with a female predominance and a peak incidence in the fifth decade . Most paBoth of these tumor types are composed of two kinds of cells. These include cuboidal epithelium lining cells and round cells with a variable architecture, including papillary, sclerotic, solid, and hemorrhagic. Molecular studies indicate that PSPs are true tumors, unlike a typical hamartoma . PSP canHerein, we report an unusual case of PH. Our patient is a young female as opposed to the common presentation of males in their fifties or sixties. The location is also unusual with an extremely rare finding of a neighboring PSP. PHs are benign, rare, and usually present as a solitary pulmonary nodule on chest radiography. Definite diagnosis and treatment can be achieved by surgical resection as it is important to exclude a possible malignancy. In addition to that, proper visualization and exploration of the surrounding area in relation to the primary lesion may help discover a second hidden lesion, as demonstrated in our case.


Tumor-associated hypoglycemia can be caused by non-islet cell tumors including gastrointestinal stromal tumor (GIST) which is a rare paraneoplastic syndrome that leads to the release of insulin-like growth factor-2 (IGF-2). We report the case of a 45-year old woman who was admitted to our hospital with refractory hypoglycemic episodes. We found normal serum insulin and c-peptide level and abdominal CT-scan showed a small duodenal wall lesion suggesting insulinoma. After tumor resection, hypoglycemia symptoms were recovered, but the pathological findings demonstrated the lesion was GIST.In a small gastrointestinal lesion with hypoglycemic symptoms we should consider IGF-II secreting GIST in addition to insulinoma. Hypoglycemia is a common event confronted in clinical practice and is usually presented simultaneously with the treatment of diabetes mellitus. This condition can be induced by insulin or other antidiabetic agents , though We report the case of a 45-year-old woman who was referred to our hospital for evaluation after some episodes of hypoglycemia neurological symptoms including weakness, fatigue, dizziness, blurred vision, headache, improving after ingestion of sweetmeat and simple carbohydrates but not including loss of consciousness, confusion, bowel habit change, and seizure. In addition, a low serum glucose level was documented in the last episode . Her symptoms began 2 months before admission and almost occurred twice a month. The episodes often occurred in fasting time and occasionally after food consumption. Her blood glucose was in the range of 40 to 70 mg/dl during hypoglycemic episodes. She had about 20kg weight loss during the last 3 months despite increased food intake and also suffered from abdominal epigastric pain. She had no significant past medical history and any medications without any point in family history. The physical examinations and vital signs had no abnormal finding. Due to persistent neuroglycopenia symptoms despite oral sweat foodstuff consumption, she was prescribed D/W 10% (Dextrose Water) serum to relief the symptoms. Her complete blood cell count, electrolytes, BUN, Cr and liver function tests were normal. Other laboratory tests are placed in According to fasting hypoglycemia and Whipple tirade , insulinoma was strongly considered. For lesion localizing, a triphasic abdominopelvic CT scan was accomplished. The finding was a 10*12mm nodular lesion in the posterior wall of the third duodenal portion anterior to psoas muscle in the level of third lumbar vertebrae which had strong enhancement in arterioportal phase . There wAll of the evaluations indicated that a duodenal wall lesion is the reason for hypoglycemia presence. Surgical treatment was scheduled. During exploratory laparotomy after Kocher maneuver, a round shape, firm consistency lesion 12mm diameter in the third portion of duodenum originating from the seromuscular layer  was founIn pathologic study, low grade gastrointestinal stromal tumor with spindle cell type and 1/50 HPF mitotic rate without necrosis was reported . In immuSymptomatic hypoglycemia which causes neurologic signs and symptoms, is called neuroglycopenic signs. These signs and symptoms are generally nonspecific, on the other hand, it is impossible to define an accurate plasma glucose level that leads to neuroglycopenia. Therefore, hypoglycemia is confirmed by documentation of the Whipple’s triad which defines symptomatic hypoglycemia, consisting of signs and symptoms of hypoglycemia, a document of low plasma glucose level and terminated signs and symptoms after increase in blood sugar level . The most prevalent etiology of hypoglycemia is alcohol abuse, using drugs affect plasma glucose level sepsis or liver dysfunction. Hypoglycemia can be also induced by different metabolic disorders . As MuraAnother uncommon etiology of hypoglycemia is tumoral lesions. The most widespread etiology is insulinoma. Other pathologies, can either induce hypoglycemia. The category of extra pancreatic tumors inducing hypoglycemia is termed as non-islet cell tumor with hypoglycemia (NICTH). NICTH is a rare paraneoplastic syndrome with an estimated incidence of nearly 1/1,000,000 person-years . NICTH iIt has been reported that GIST can rarely represent hypoglycemic episodes which this feature is a rare manifestation . These cAs this case presented some rare clinical findings, it makes it unique. These findings are extra ordinal when we compare our case with the patients suffering from hypoglycemia inducing GIST that were previously reported , 4. ThisIn our case, fasting hypoglycemic episodes terminated immediately after surgery and no longer hypoglycemic episodes occurred, on the other hand, our investigations showed a small single lesion leading to hypoglycemia, but in literature, this was in contrast with other reports as most of the studies reported a metastatic or a large tumor causing hypoglycemia. , 11. As It is very difficult to explain the normal serum insulin level and hypoglycemia in this case based on mechanism of IGF, c-peptide, and insulin as unfortunately we did not check the IGF-II level. We think that hypoglycemia along with normal insulin levels might be due to the clinical stage, since our patient stood in the initial stages of GIST and almost all previously reported patients were in metastatic stages , 4. HoweIn conclusion Hypoglycemia inducing GIST is a type of NICTH which is reported rarely. The leading mechanisms include insulin-like growth factor II hypersecretion. The hypoglycemic symptoms often are presented in huge or metastatic GIST, but in our case, a single small GIST causes the symptoms. It was cured by resection. Thus in a small GI lesion with hypoglycemic symptoms, we should consider IGF-II secreting GIST in addition to insulinoma.


Acute appendicitis is one of the most common surgical diagnoses in clinical practice. In case of uncomplicated course, diagnosis and treatment do not cause significant difficulties. On the other hand, unrecognized or complicated appendicitis can rarely bring unusual complications that threaten the patient with delayed treatment rather than the course itself. Portal vein thrombosis, also known as pylephlebitis, with an incidence of 1/1000 acute admissions, certainly meets this statement.In this study, we present a successful treatment of advanced acute appendicitis complicating treatment of biliary obstruction. Due to the advanced inflammation with forced intestinal resection in the extent of right-sided hemicolectomy. And then successful conservative treatment of portal vein thrombosis in the surgical facility lasting a total of 6 weeks when the patient was discharged to home care without sequelae.The epidemiology, presentation, diagnosis and strategy of treatments as well as their outcomes were discussed.Portal vein thrombosis after acute appendicitis is rare. In case of unfavorable postoperative course with high inflammatory markers, temperatures, discomfort and abdominal pain, a CT scan is in order, which can easily establish the diagnosis and subsequently target the treatment in the right direction. Treatment of pylephlebitis is conservative and long term. It consists in the application of low molecular weight heparin and targeted antibiotic treatment. The mortality rate is 32%. •Appendicitis following cholangoitis•Right hemicolectomy•Pylephlebitis•Treatment However, rare complications are described in the literature, occurring in units of cases and at different intervals from the procedure, which may lead to diagnostic confusion, as well as their treatment may not be standard. Vascular complications involving both intestinal necrosis and venous thrombosis have been described. Inflammatory complications range from inflammation of the abdominal wall to variously localized abscesses. And finally, perhaps not so rare, stump appendicitis. Advanced appendicitis causing intestinal necrosis on account of small vein thrombosis may not in itself impose a significant burden on the patient, perhaps only complications arising from intestinal resection. However, the ascending thrombosis of the portal vein, either due to appendicitis or cholangitis, is already a certain complication, moreover with significant morbidity. Despite treatment, it is fatal in up to 32% of cases and has a significantly worse prognosis if left untreated. Only a few cases of predominantly pylephlebitis following acute appendicitis have been described in the available literature. In this report, we describe a case of a patient with concomitant cholangitis, appendicitis, and portal vein thrombosis. This paper has been reported in line with the SCARE 2020 criteria 2We present the case of a 49-year-old male patient, a non-smoker with a BMI of 33, a history of knee arthroscopy, and treated for hypothyroidism. No family burden was noted. The patient was seen in the emergency department of a university hospital for right subcostal pain spreading to the back and epigastrium after a dietary mistake. Among other complaints, he reported a temperature with a maximum of 37.5st C.Laboratory showed only mild inflammation . Ultrasound and subsequent CT scan confirmed bile duct dilatation with no evidence of other pathology in the abdominal cavity. The patient was admitted to the gastroenterology department and underwent ERCP examination with papillosphincterotomy and biliary sludge extraction. The procedure was performed by an expert endoscopist with 20 years of experience and well-tolerated by the patient. In the following course, despite antibiotic therapy, the clinical condition deteriorated. Therefore, a follow-up CT scan was performed four days after the procedure and described advanced appendicitis with subhepatically located appendix. Proteus mirabilis was repeatedly cultivated. However, the condition was still not favorable, a follow-up postoperative CT scan 5 days apart revealed an intra-abdominal abscess which was drained under CT control, and portal vein thrombosis which was treated conservatively. Targeted antibiotic therapy was administered, 3Proteus mirabilis, Klebsiella pneumoniae, anaerobic streptococci, and clostridia In general, complications can be divided into early and late. Leaving aside the common ones such as an abscess or surgical wound infection, thrombosis of the ileocolic or portal vein associated with intestinal necrosis is rarely observed. Pylephlebitis or portal vein thrombosis is a rare intra-abdominal vascular complication. The incidence is reported to be 1/1000 acute admissions Treatment of pylephlebitis is mainly antibiotics and anticoagulation. Primarily empirical curative dose of antibiotics, which is subsequently adjusted according to the cultivation result, as well as a therapeutic dose of low molecular weight heparin as early as the postoperative condition allows. Treatment is long-term, antibiotic 4–6 weeks, anticoagulant 3–6 months 4Acute cholangitis or appendicitis does not seem to be a major complication of routine medical practice, this statement is especially true for an uncomplicated course. However, problems, both diagnostic and therapeutic, may arise in the case of an atypical course or coexisting disease, as well as in the presence of rare complications. The insidiousness, incidence, and mortality of pyelophlebitis are certainly worth mentioning. It is also worthy of our attention, especially in identifying the cause of atypical postoperative course.Not commissioned, externally peer-reviewed.10.13039/501100003243Ministry of Health, Czech Republic – conceptual development of research organization .Supported by Not required in our institution to publish anonymous case reports.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.MUDr Tomáš Řezáč Ph.D.Tomáš Řezáč: design of the study, collection of the data, drafting the manuscript, final approval of the version to be submitted.Pavel Zbořil: revising the manuscript, final approval of the version to be submitted.Katherine Vomáčková: revising the manuscript, english correction, final approval of the version to be submitted.Petr Špička: revising the manuscript, final approval of the version to be submitted.The authors declare that they have no conflict of interest related to individual authors' commitments.The authors declare no potential conflicts of interest related to commitments of editors, journal staff, or reviewers.


Acute gastric volvulus is an uncommon emergency department (ED) presentation associated with high mortality from gastric ischemia and perforation. The diagnosis of this pathology is complicated by its intermittent symptoms and similarity in presentation to more common disorders encountered in the ED. Assessing for key risk factors, such as the presence of a hiatal hernia, and the use of expeditious imaging modalities, such as bedside radiography and point-of-care ultrasonography, are essential in rapid diagnosis and time-sensitive, definitive surgical intervention. Gastric volvulus is a rare, potentially life-threatening pathology that occurs when the stomach is abnormally rotated along its long or short axis . This roIn the emergency department (ED), the diagnosis of gastric volvulus is difficult due to its rarity and similar symptomatology to more common ED presentations in both the acute and chronic forms of the disease. For example, acute presentations may be confused with pancreatitis and acute coronary syndrome, whereas intermittent, chronic presentations may be falsely attributed to peptic ulcer disease, gallbladder pathologies, and gastroesophageal reflux disease ,5. Here,A 75-year-old woman with a past medical history of hypertension, hyperlipidemia, stroke without residual deficits, appendectomy, laparoscopic repair of a hiatal hernia two years prior, and an anaphylactic allergy to contrast dye presented to the ED with left-sided abdominal pain that started one day prior to arrival. She reported that the pain started on the left side of her left abdomen and then progressed to include the right side of her abdomen. Her pain was associated with retching and vomiting that was non-bilious, non-bloody, and progressed to dry heaving. The patient reported her last bowel movement occurred on the morning prior to arrival and was normal. Notably, she had a milder episode of similar symptoms about 6 months ago; however, she elected not to present to the hospital because the pain subsided. She denied a history of recent trauma, coronary artery disease, and use of any blood thinner mediations.9/L, hemoglobin/hematocrit of 10.1/31.7, and platelet count of 226 x109/L. Her lactic acid and lipase levels were 2.0 mmol/L and 85 U/L, respectively. The electrocardiogram, troponin, electrolyte, and hepatic tests were unremarkable.On her initial vitals, the patient had pulse oximetry of 98% on room air, blood pressure of 106/58 mmHg, temperature of 98°F, respiratory rate of 18, and heart rate of 86. The patient was in no apparent distress. She had a kyphotic habitus, a regular rate and rhythm, clear lung sounds, and normal pulses in all extremities. On her abdominal exam, she was non-distended with upper abdominal rigidity and severe tenderness to palpation. Laboratory evaluation revealed a white blood cell count of 7.0 x10On initial imaging, bedside chest radiography was remarkable for a moderate size hiatal hernia, left basilar density with blunting of the left costophrenic angle, eventration of the right hemidiaphragm, and scoliosis Figure .Point-of-care ultrasound (POCUS) was remarkable for free fluid most prominent in the suprapubic window. The surgical team was immediately consulted and the patient was administered broad-spectrum antibiotics and intravenous (IV) fluid resuscitation. Given the patient’s reported history of anaphylactic allergy to contrast dye, the patient was pre-medicated with IV methylprednisolone and diphenhydramine prior to computed tomography (CT) imaging. CT with contrast angiography was remarkable for a large hiatal hernia with suspected organo-axial volvulus and free fluid surrounding the distal esophagus with moderate hemoperitoneum surrounding the stomach, liver, spleen, and deep pelvis without evidence of active arterial extravasation Figures -4.The patient was immediately taken to the operating room for NGT placement and exploratory laparotomy. The patient was found to have about 2 L of hemoperitoneum and a paraesophageal hernia recurrence, which was reduced and repaired with mesh, and a gastric volvulus, which was repaired with gastropexy. The patient was transferred to the intensive care unit for post-operative care and was ultimately downgraded with a good prognosis for recovery.We report a case of an acute gastric volvulus in the setting of a paraesophageal hernia with hemoperitoneum. Our case was complicated by its likely early presentation and rapid progression of acute on chronic gastric volvulus as underscored by the patient’s recent bowel movement and unremarkable initial laboratory values. Importantly, our patient possessed multiple key risk factors for a gastric volvulus, including an age >50, a history of a paraesophageal hernia, diaphragmatic eventration on chest radiography , and kyphoscoliotic anatomy. General risk factors include a history of diaphragmatic anomalies and any hiatal hernia , phrenic nerve paralysis, and other anatomic stomach or splenic abnormalities . The patGastric volvuli have multiple modes of rotation that are associated with different presentations Figure .In our case, the patient’s volvulus was organo-axial in rotation, whereby the stomach rotates around its long axis along the gastroesophageal junction and the pylorus, where the antrum rotates anterosuperiorly and the fundus rotates posteroinferiorly resulting in an inverted position . The orgMultiple imaging modalities may be employed in the diagnosis of gastric volvulus. The gold standard test is a barium swallow, which has very high sensitivity and specificity for diagnosing a gastric volvulus; however, this modality may not be feasible in the ED setting . In contIn the ED, the initial approach to patients with upper abdominal pain and vomiting includes stabilization and simultaneous assessment of a wide array of life-threatening etiologies. In particular, acute coronary syndrome, aortic dissection, and abdominal pathologies including acute mesenteric ischemia, aortic rupture, gastrointestinal perforation, and bowel obstruction can each present similarly and involve different intervention pathways. Given the wide differential diagnosis, the use of rapid imaging modalities such as portable chest radiography and POCUS is essential in narrowing the differential and rapid mobilization of surgical resources in preparation for definitive care. Once the diagnosis of gastric volvulus is made, placement of an NGT is an option in cooperative patients for gastric decompression in order to reduce stomach tension, which may improve perfusion and decrease the risk for gastric ischemia and necrosis . In addiAcute gastric volvulus is associated with high mortality from gastric ischemia and perforation. In the ED, narrowing down the wide differential diagnosis associated with the nonspecific presentation of gastric volvulus is dependent on an astute assessment of risk factors and clinical examination as well as rapid imaging modalities, such as bedside radiography and POCUS. Efficient diagnosis and immediate surgical consultation are essential in facilitating time-sensitive, definitive surgical intervention.


Hepatic inflammatory pseudotumor (IPT) is a rare, benign, tumor-like lesion. Because there are no characteristic laboratory markers or radiological features, hepatic IPT is often misdiagnosed as a malignant neoplasm such as hepatocellular carcinoma (HCC).Lens culinaris agglutinin-reactive fraction, AFP lectin 3 (AFP-L3), were elevated to 822.8 ng/ml and 75.2%, respectively. The tumor showed contrast enhancement on contrast-enhanced computed tomography and various accumulation on positron emission tomography. Based on these biological and imaging features, HCC was suspected, and we performed laparoscopic partial hepatectomy for these two tumors. Pathological diagnosis revealed that both tumors were hepatic IPTs with no malignant characteristics. After hepatectomy, the serum AFP and AFP-L3 levels decreased to the normal range.A 68-year-old man with liver dysfunction due to chronic hepatitis C virus infection and alcoholic liver disease presented with hepatic tumors in segments III and VIII. The levels of serum alpha-fetoprotein (AFP) and its We report a very rare case of hepatic IPT with elevated serum AFP and AFP-L3, mimicking HCC. Clinicians should include this rare neoplasm in the differential diagnoses of hepatic tumors even when the serum markers for HCC are elevated. Lens culinaris agglutinin-reactive fraction AFP lectin 3 (AFP-L3), and radiological findings.Hepatic inflammatory pseudotumor (IPT) is a rare benign tumor-like lesion that was first described in 1953 by Pack and Baker , 3. TherA 68-year-old man, who had been undergoing medical treatment for liver dysfunction due to chronic hepatitis C virus (HCV) infection and alcoholic liver disease, was found to have a liver tumor measuring 25 mm in segment III on abdominal ultrasonography and was referred to our hospital for further evaluation. He was on antihypertensive medication and had undergone open reduction and internal fixation with metal plates for the fracture of his left upper extremity. He was a past smoker, with a daily alcohol intake of 80 g for 42 years. Laboratory analysis revealed the following results: aspartate aminotransferase, 46 U/L ; alanine aminotransferase, 51 U/L ; alkaline phosphatase, 210 U/L ; and gamma-guanosine triphosphate, 27 U/L . Serum AFP and AFP-L3 were 822.8 ng/mL  and 75.2% , respectively. Levels of protein induced by vitamin K absence or antagonist II, carcinoembryonic antigen, and carbohydrate antigen 19-9 were within normal limits. Serological tests for hepatitis viruses showed that hepatitis B surface antigen was negative, but hepatitis B surface antibody, hepatitis B core antibody, and HCV antibody were positive. There was no anemia or elevation of white blood cell count and C-reactive protein level. Coagulation profile, total protein, albumin, and bilirubin levels  were normal. Contrast-enhanced computed tomography (CT) showed a 24-mm mass in segment III of the liver adjacent to the left hepatic vein, and an 8 mm mass in segment VIII. These lesions showed weak enhancement in the early phase and demonstrated washout of the contrast in the delayed phase (Fig. Macroscopically, the resected specimen showed hard white nodules, not like HCC, in approximately the same location that was indicated by the imaging studies Fig. . HistopaThe patient had no postoperative complications and was discharged 7 days after surgery. AFP and AFP-L3 levels decreased to 13 ng/mL and 38.9%, respectively, 1 week after of surgery, and returned to normal levels 3 months later, to 5.5 ng/mL and < 0.5%, respectively. These tumor markers were not elevated during the 1-year follow-up.In the present case, the preoperative diagnosis was HCC because of the patient’s background chronic liver disease, elevated serum AFP and AFP-L3, and radiological findings such as high–low pattern. In hepatic IPTs, although inflammatory markers, including C-reactive protein and leukocyte count, and liver enzymes are sometimes elevated, tumor markers are usually normal . AFP is Radiological findings of IPT are also non-specific. CT scan often shows a low-density mass , 9–11. OOnce a definitive diagnosis is obtained, conservative management is the treatment of choice for IPT. However, as mentioned above, differentiating IPT from malignancy, on the basis of noninvasive diagnostic methods, is very difficult. In fact, surgical resection is frequently selected as the treatment of choice for hepatic IPTs because these tumors were suspected to be malignant tumors, preoperatively, in many cases , 11, 15.In conclusion, we encountered a rare case of multiple hepatic IPTs with elevated AFP and AFP-L3 levels, mimicking HCC. We should keep in mind that even if these tumor markers, especially AFP-L3, are elevated, it does not always indicate the presence of HCC, as in our reported case. Our report also shows that hepatic IPT can be one of the benign tumors associated with elevated AFP and AFP-L3 levels.


We report a case of subacute rebound iritis characterized by a globular, pedunculated anterior chamber mass that resolved after topical steroid burst.A 59-year-old female with no significant past ocular history underwent cataract extraction (CE) with phacoemulsification and posterior chamber intraocular lens (PCIOL) insertion in the right eye (OD). Intraoperatively, a Malyugin ring was deployed due to poor dilation, however the remainder of the surgery was uneventful. On the first postoperative day, 3+ mixed cell and pigment in the anterior chamber (AC) without fibrin reaction was observed, which resolved by the second week with topical steroid administration. The patient was lost to follow up and was non-compliant with her steroid taper.Eight weeks later, the patient presented with eye pain OD. Visual acuity (VA) was 20/40 and intraocular pressure (IOP) by Goldmann applanation was 6; there was no relative afferent pupillary defect (rAPD) noted. Slit lamp biomicroscopy was remarkable for trace Descemet’s folds (DF), 3+ mixed AC cell and pigment, and a sharply-circumscribed, globular, partially opaque anterior chamber mass with smooth borders and a well-demarcated stalk attached to the surface of the PCIOL Fig. A and B. The patient was started on prednisolone acetate 1% eye drops every two hours and cyclopentolate 1% eye drops twice a day. Examination three days later showed complete resolution of the mass Fig. A and B aThe post-operative anterior segment mass reported above most likely represents a fibrinous exudate secondary to rebound iritis. Rebound and persistent iritis are well-known entities that may occur after cataract surgery. Neatrour et al  reportedFibrinous exudates are occasionally encountered after intraocular surgery, more commonly after pars plana vitrectomy –5. FibriThe pathophysiology of post-operative fibrin clots is thought to be secondary to a transient lowering of IOP and disruption in the blood-aqueous barrier during CE, resulting in leakage of fibrinogen-rich fluid from arterial plasma into the AC, eliciting a fibrinoid reaction –9. If unTo the best of our knowledge, this is the first report in the ophthalmic literature of a subacute post-operative anterior uveitic mass in such a well-circumscribed configuration that completely resolved after a short course of topical steroids. We surmise that this mass represents a fibrinous exudate in the setting of rebound iritis after cataract surgery involving a pupil expansion device. Ophthalmologists should be aware of this unique presentation after intraocular surgery.


A MEST is a rare renal tumor, with stromal as well as epithelial components. It is predominantly benign and local recurrence is not very common. In the majority of the cases, it occurs in females. Its occurrence in a young male makes it a rarity.A 24 years old male presented at SIUT with the complaint of left flank pain on and off for one month. CT scan showed soft tissue density mass in left renal pelvis extending from mid-pole calyces to pelviuretric junction, leading to obstruction and ultimately mild uropathy. We found a partially obstructing staghorn calculus with asymmetrical cortical thinning. Left Robot-Assisted Nephro-ureterectomy plus excision of bladder cuff was planned in which 3 × 4 cm mass involving the left renal pelvis was excised. To date, there is no radiologic evidence of disease recurrence.MEST in young adults is an extremely rare tumor. They have been referred to by many alternate synonyms including ‘adult mesoblastic nephroma’ and ‘cystic nephroma’ with ‘ovarian’ or ‘cellular’ type stroma. Majority of patients with MEST present, with hematuria, abdominal pain, palpable flank mass, recurrent urinary tract infections. Similarly, our patient presented initially with nonspecific pain in the left flank region. Majority of cases in the literature presented with the tumor in benign stage, with localized spread, and without recurrence.Mixed epithelial and stromal tumors (MEST) of the kidney are distinct entities of benign kidney tumors. MEST in young males is a very rare entity, and a small number of cases exist. Histopathology plays a very cardinal role in diagnosis, and overall the disease has a promising outcome with conservative surgery. •There are only very few cases exist in literature on MEST in young males which makes it an extremely rare entity.•In the recent WHO classification of renal neoplasms, cystic nephroma was recognized as a separate entity, with MEST being tumors composed of a mixture of cysts, microcysts and tubules with variable stroma of cellular type.•Histopathology plays a very cardinal role in diagnosis.•Overall the disease has a favorable prognosis with conservative surgery. The stromal element resembles the ovarian stroma and chiefly entails the spindle cells, which contain progesterone and estrogen receptors on their surface. Epithelium-lined cysts or micro cysts mainly make up its epithelial component . In the This case report has been reported in line with the SCARE Criteria .2A 24 years old, unmarried male, resident of Dera Ghazi Khan, presented at Sindh Institute of Urology and Transplantation (SIUT) with the complaint of left flank pain on and off for one month. He had a positive family history of stone disease. He had a history of stone passage and appendectomy being attempted in 2007. On examination, he was a young male of average height and built, well oriented to time, place, and person. Hematological investigation showed hemoglobin of 11.2g/dl, platelet count 280000 × 109 per liter, Total leucocyte Count (TLC) 5.2 × 109/L. Urine D/R and Urine culture were negative. Serum Creatinine was 0.88mg/dl, blood urea nitrogen was 18 and an international normalized ratio (INR) of 1.01 was found. Serologic tests for hepatitis B virus, hepatitis C, and human immunodeficiency virus (HIV) were negative. Vitals included pulse 74/min, afebrile, respiratory rate 16/min, and blood pressure was 136/78 mmHg. On General physical exam Anemia and Jaundice were negative, Lymph nodes were not palpable, and Abdomen was soft and non-tender. A Healed scar mark in the right iliac fossa was noted. No swelling was appreciated on the cough. The rest of the examination was unremarkable. Bladder histopathology report showed mild, chronic, non-specific cystitis. No evidence of dysplasia, TB, or tumor was seen. Cystoscopy and biopsy were done which showed no remarkable findings.A CT scan was done in Sindh Institute of Urology and Transplantation (SIUT) that revealed soft tissue density mass in the left renal pelvis extending down to mid-pole calyces to pleviuretric junction leading to left renal pelvis distention and obstruction. This obstruction resulted in mild to moderate uropathy on the same side  is a rare, adult, biphasic tumor of the kidney that has an admixture of an epithelial and mesenchymal component. It was first recognized as a distinct entity by Adsay et. Al and Michal and Syrucek et. Al in 1998 ,12. TheyMajority of patients with MEST present, with urinary symptoms such as hematuria, abdominal pain, palpable flank mass, recurrent urinary tract infections, or simply as an incidental finding. A review of the literature reveals more cases presenting as incidental findings which could be explained due to advanced radiological investigations and increasing awareness amongst patients for medical checkups, both routine and non-routine . Our patMajority of cases in the literature presented with the tumor in benign stage, with localized spread, and without recurrence . PresentOn gross examination, the tumor in most cases reveals a variably circumscribed, encapsulated tan yellow with an admixture of solid and cystic areas, either of which may be more dominant . In our On microscopy, the tumor consists of epithelial and stromal components. The mesenchymal component presents as fascicles of spindle cells with smooth muscle, fibroblastic, and/or myofibroblastic tissue interspersed with bundles of collagen. The epithelial component is an important part of the tumor, and is not limited to the surface, and may be found within the mesenchymal tissue. In our case, the tumor showed tubules with cysts lined by columnar cells demonstrating hob nailing pattern, consistent with literature which also has evidence of simple to complex tubule-papillary pattern with or without dilatation by cysts . MitosesOur patient also had a significant history of the passage of stones 13 years back, with a strong family history. During current management, the CT scan abdomen initially revealed two large calculi in the lower pole of a left kidney that progressed to partial staghorn calculi in the next CT scan. The calculi may be contributing to the hydronephrosis and resultant abdominal pain. Such calculi were not observed in literature so far, making this a unique finding. Jing Ye et al. proposed that MRI is a better investigation to diagnose this condition in his article, along with imaging guidelines for this particular tumor on account of its rarity . HoweverIn immunohistochemistry, the mesenchymal component shows smooth muscle differentiation as in our case, and the epithelial component-in majority of cases-shows positive estrogen and progesterone receptors, a finding strongly found in our patient after removal of tissue and study . CytogenDifferential diagnoses of tumors that may present as MEST include cystic nephroma, as they both share multiple clinical, morphologic, histopathological, and immunohistochemical features, and Turbiner et al. proposed both of them to be unified into a single entity titled “renal epithelial and stromal tumor (REST)” .4In conclusion, mixed epithelial and stromal tumors of the kidney are a distinct entity of benign kidney tumors, which should be placed down on the differentials of a patient presenting with non-specific abdominal and/or genitourinary symptoms. MEST in young males is an extremely rare entity, and very few cases exist in literature. Histopathology plays a very cardinal role in diagnosis, and overall the disease has a favorable prognosis with conservative surgery.The patient did not present his point of view.Not commissioned, externally peer reviewed.This is a case report that does not require a formal ethical committee approval.The authors declare that they have no funding.MTT, KSK: Study concept or design; MTT, SU, KSK, AK, IU: Data collection, data analysis or interpretation, writing the paper and IU: Critical revision of the article.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.This is a case report that does not require a registration of research studies.Kiran Shafiq Khan and Irfan Ullah.The authors declare that there is no conflict of interest.


Primary dural lymphoma (PDL) usually arises from the calvarial dura without the brain parenchyma or systemic involvement and thus may not be considered as a typical form of primary CNS lymphoma (PCNSL). It is exceedingly rare. When it occurs, it might not be suspected as a primary diagnosis on clinical and radiologic findings. We present a PDL case that occurs at the cerebellopontine (CP) angle mimicking en plaque meningioma. The tumor histopathology showed a lymphoproliferative disorder immunophenotypically consistent with a low-grade marginal zone lymphoma. Bone marrow and systemic involvements were not identified, and a diagnosis of PDL was established. As a residual tumor at the CP angle was inaccessible to surgery, postoperative radiation therapy was performed. No recurrence was found at 15-month follow-up. PDLs are mostly indolent and have a good prognosis. There is no doubt that the most important differential diagnosis is meningioma. Furthermore, the present case emphasizes the necessity of an intraoperative consultation and knowledge of this rare yet essential form of PCNSL so that appropriate studies can be ordered. PCNSL is an extranodal non-Hodgkin lymphoma that commonly involves the brain, spinal cord, leptomeninges, and ocular structures . Dura inA 67-year-old female patient was initially seen for left-sided facial and mouth numbness, watering eye, and altered taste, which progressed over one year. MRI of the brain with and without contrast revealed a 2.6 × 2.1 cm well-defined homogeneously enhancing extra-axial dural-based mass at the left CP angle, which extended superiorly to the tentorium, suspicious for en plaque meningioma . The patPDLs grow from the dura without the brain parenchymal or systemic involvement. The lymphomas infiltrate meningeal tissue, which often presents with sclerotic features. Most of PDLs arise from the calvarial dura. According to Karschnia et al., 55% are from the calvarial dura, 30% from the skull base, and 15% from the spinal dura out of 20 PDL cases in the database . They rePDL is rare and usually has a different prognosis from PCNSL. Surgical excision has been established as the standard treatment for the past decades. However, complete resection could be a challenge if PDL presents as an infiltrative and/or en plaque lesion. Adjuvant treatment with either radiation or chemotherapy is then indicated. In this case, postoperative external beam radiation to the residual tumor allowed complete remission. Since recurrence can develop after complete remission following the initial treatment, regular follow-up is highly recommended . Althoug


The use of whole-genome sequencing in clinical practice has revealed variable genomic characteristics across cancer types, one of which is whole-genome doubling (WGD), which describes the duplication of a complete set of chromosomes. Yet it is relatively rare in prostate cancer and no such case has ever been reported in Japanese patients.A 54-year-old patient with prostatic adenocarcinoma with bone and lymph node metastases was started on androgen-deprivation therapy. As the prostate cancer turned castration-resistant, multimodal therapies including taxane- and platinum-based chemotherapy, androgen-receptor antagonist inhibitors, radiotherapy and radium-233 were introduced. Good controls of serum prostate-specific antigen (PSA) level and bone metastases were achieved for more than 13 years since after the initial treatment. During the treatment, a metastatic lymph node biopsy was performed to confirm the tumor histology, and spinal decompression surgery were performed for spinal compression due to lumber vertebral metastases. The immunohistochemical analysis identified PSA and androgen receptor positive tumor cells in both metastatic lesions, while no variable cancer cells were detected in the prostate on second biopsy. Whole-genome sequencing was performed on the biopsied metastatic lymph node in search for another possible treatment and it revealed that the tumor had WGD and CDK12 mutation. The WGD-positive tumor cells contained large and polymorphic nucleus, presumably reflecting on the ploidy abnormality of the chromosomes.This report is the first case of a Japanese patient presenting with WGD, who survived more than 13 years with multimodal chemotherapies and radiotherapies.The online version contains supplementary material available at 10.1186/s12920-022-01178-z. Recently large-scale genomic studies have enabled utilization of whole exome-sequencing (WES), the sequencing of all the protein-coding regions of individual cancer-patient genome, in search for precision therapeutic strategies. Major driver-gene mutations found in this the WES could provide candidates for applicable therapeutic medicine beyond cancer types . ApplicaA 54-year-old male with an elevated serum prostate-specific antigen (PSA) level (573.9 ng/mL) was referred to our facility. He had no remarkable medical or family histories. Transrectal prostate biopsy revealed prostate cancer in 9 out of 10 cores, with a Gleason score of 4 + 5. Through MRI and computed tomography (CT), a tumor was detected in the right lobe of the prostate, clearly expanding beyond the capsule to the right seminal vesicle; multiple lymphadenopathy and bone metastases were also noted. Thus, he was diagnosed with cT3bN1M1b prostate cancer. At the first diagnosis of prostate cancer, the patient was started on androgen deprivation therapy (ADT) with luteinizing hormone-releasing hormone (LHRH) agonist in combination with antiandrogens such as bicalutamide, flutamide, ethinylestradiol as the first-line therapy, which dramatically decreased his PSA level to a nadir of 0.02 ng/mL after 19 months. Although PSA relapse was observed at 31 months, his serum PSA level was maintained below 2.0 ng/mL with ethinylestradiol for 27 months. However, as shown in Fig. We started docetaxel (75 mg/m2) as the second-line therapy, which decreased his PSA level to 1.70 ng/mL after six cycles. However, CT scan showed metastatic progression in the right pelvic bone and new metastases in the sacrum, pubis, and right lung. As the third-line therapy, enzalutamide was administered, but the treatment was halted after 7 months because it failed to decrease the patient’s PSA level. Thus, as the fourth-line therapy, 25 mg/m2 of cabazitaxel (CBZ) was introduced, and it initially decreased his PSA level to 4.86 ng/mL. However, PSA relapse was observed after 4 months and radium-223 (Ra-223) was added to the regimen. As shown in Fig. Second prostate biopsy was performed 132 months after the primary diagnosis in search for possible neuroendocrine differentiation, which was suspected because of the high serum neuron-specific enolase (NSE) level of 30.7 ng/mL. Conversely, no viable tumor cells were detected in the primary lesion. Instead of CBZ, abiraterone was introduced as the fifth-line therapy, and radiation therapy (37.5 Gy/15 Fr) was given to the pelvic bone metastases, leading to a dramatically decreased PSA level from 81.77 to 19.9 ng/mL. However, the patient’s PSA level rose again after 6 months, and new metastases in the L4/5 vertebrae were detected in the bone scan. Based on the increased serum NSE level (30.9 ng/mL), we again suspected neuroendocrine differentiation and decided to introduce the sixth-line therapy comprising of an 80% dose of cisplatin (CDDP) and a 100% dose of etoposide (VP-16) along with radiation therapy to the L5 vertebrae, sacrum, left pubis, and right femur, which resulted in decreased PSA and NSE levels. Although bone scans revealed decreased accumulation in the bone metastases, the patient’s PSA level began to increase 11 months after the initiation of CDDP + VP-16 therapy. Four cycles of CBZ were administered as the seventh-line therapy, and four cycles of carboplatin (CBDCA) and irinotecan (CPT-11) were continuously introduced as the eighth-line therapy. However, the patient’s PSA level further increased, and expansion of lymph node metastases was detected on CT scan.We performed a CT-guided biopsy on the swollen right inguinal lymph node to study the tumor’s pathological characteristics Fig. . The bioSeeking other possible therapies, the patient agreed to undergo whole-exome sequencing (WES) using the PleSSision Exome test and samples were collected via lymph node biopsy. WES through the PleSSision Exome test analyzes both tumor and germline DNA. As it is a tumor-normal matched paired test, genomic deoxyribonucleic acid (DNA) was extracted and purified from tumor tissues and from peripheral blood mononuclear cells obtained from the patient. This examination can detect both single-nucleotide variants (SNVs) and copy number variants (CNVs) of 19,296 genes, enabling a wide range of diagnoses. The tumor mutation burden (TMB) is also calculated via WES by the number of SNVs, insertions, and deletions per mega-base in coding regions. The analysis revealed mutations in CDK12 (p.G101Dfs*23), FOXA1 (p.R261C), MACC1 p.M480L), and HIP1 (p.Q265E). There were no mutations or amplifications of AR, TP53, PTEN, SPOP, MYC, or PI3K pathway and WNT pathway genes. DNA repair genes except for CDK12 had no alterations. Copy number amplifications were observed in 85 driver genes, including CDK12. Detailed information about gene alterations is presented in Additional file 80L, and We used OLYMPUS IX83 (microscope) and OLYMPYS UPLFLN10X, UPLFLN20X (objective lens) and OLYMPUS DP80 (CCD camera) for histological analysis of the specimens. The images were captured via OLYMPUS cellSens Dimension 1.14. The resolutions of the original images were all 72 dpi, while the resolutions of processed images shown in the Fig. Since the mechanism of the AR axis was revealed, metastatic castration-resistant prostate cancer (CRPC) has been conventionally treated with AR pathway inhibitors such as abiraterone and enzalutamide, or taxane-based chemotherapies. Recently, a new treatment approach using whole-exome sequencing for advanced prostate cancer has been initiated. In this analysis, approximately 20,000 of all the genes of the human genome were surveyed to detect clinically important gene mutations and the status of TMB and MSI . In addiIn the present case, WES revealed a relatively high TMB, CDK12 mutation, and the presence of WGD. The definition of WGD is varied in previous reports, while Peter Priestley et al. defined WGD as a major allele ploidy greater than 1.5 on 50% of at least 11 autosomes as the number of duplicated autosomes . A previThe histological characteristics of the metastatic tumor in our case were unique. Many of the tumor cells contained relatively large, polymorphic nucleus, as shown in Fig. CDK12 is a cyclin-dependent kinase that contributes to DNA repair by regulating homologous recombination DNA repair genes such as ATR, BRACA1, FANCI, and FANCD2. CDK12 mutation occurs in 4%-11% of prostate cancer cases , and sevAnother interesting aspect of our case is that the patient had survived for 8 years with maintained performance status after CRPC diagnosis, probably because of the successful local therapy. While Ra-223 contributed to better OS in patients with mCRPC as shown in ALSYMPCA study , the relIn conclusion, this case report presents the first Japanese case of metastatic CRPC that underwent WGD after a series of taxane- and platinum-based chemotherapy. Although the clinical significance of WGD itself in prostate cancer remains uncertain, further development in genome sequencing may reveal its importance and clinical application.Additional file 1: Table S1. Detailed information about gene alterations in COSMIC Cancer Gene Census Tier1 or Tier2 (728 genes).


First described in 1955 Primary mediastinal seminomas are rare. Only 1–4% of mediastinal tumours are germ cell tumors; majority of which are teratomas. They typically present in men aged between 20 and 40 years. Very few cases are reported in the literature. Florid follicular lymphoid hyperplasia can obscure the malignant cells and is a rarer finding still. We present a rare case of a 48 year old man with a primary mediastinal seminoma with florid follicular lymphoid hyperplasia; found following excision of a clinically presumed thymoma.A 48 year old man was referred for excision of a thymic mass. The presumed diagnosis was a thymoma; following preoperative investigations. The mass was incidentally found on a radiological imaging. However, the patient did report mid-sternal discomfort on lying flat and breathlessness. The patient underwent a thymectomy via a partial median sternotomy with good recovery. Histological assessment was that the mass was in fact a primary mediastinal seminoma with florid follicular lymphoid hyperplasia. A primary testicular malignancy was excluded and the patient required no further oncological treatment.Only 11 cases have previously been reported of primary mediastinal seminoma with florid follicular lymphoid hyperplasia. Although rare, a primary mediastinal seminoma should be considered as a differential diagnosis for presentations with a thymic mass. Tumour markers can be helpful, however are only positive in third of cases. Ultrasound imaging of the gonads is essential to exclude a primary gonadal lesion. Pure seminomas are radiotherapy and chemotherapy sensitive however the mainstay treatment of primary mediastinal seminomas remains surgical excision. Radiotherapy is reserved postoperatively for incomplete surgical margins. Seminomas are a type of germ cell tumour that usually occur in the reproductive organs. Extragonadal germ cell tumours, on the other hand, typically present in the midline structures, such as the retroperitoneum and mediastinum –3. PrimaHistologically, primary mediastinal seminomas with florid follicular hyperplasia are even rarer. At times, secondary accompanying features such as florid lymphoid hyperplasia or cystic changes can be quite pronounced, thereby obscuring the malignant cells. Given that these are morphologically indistinguishable from their gonadal counterparts, it is therefore prudent to exclude the possibility of seminoma metastasising to the mediastinum from the gonad .3., often asymptomatic and found incidentally in 20–30% of cases. Our case is of a 48 year old gentleman with an incidental finding of a primary mediastinal seminoma with florid follicular lymphoid hyperplasia who had undergone an excision of a clinically presumed thymoma.In general, primary mediastinal seminomas have a good prognosis , 3, 6, 7A 48 year old gentleman was referred for excision of a thymic mass after multidisciplinary team (MDT) discussion. The mediastinal mass was incidentally found via investigation for abdominal pain which then led to Computed tomography (CT) of the chest. Retrospectively, a CT coronary angiogram from 3 years prior also showed the mass; which was not reported at the time. The mass was relatively unchanged in appearance on the latest CT. The patient reported symptoms of mid-sternal discomfort when lying flat associated with breathlessness. There were no symptoms or signs of myasthenia gravis.He had a past medical history of raised body mass index (BMI) and previous right inguinal hernia repair but was not on any regular medication. He was an ex- smoker, works as a window cleaner and lives with his wife and child. Examination was unremarkable apart from a BMI of 48.7.CT imaging, from 5 month prior to his operation, showed a slightly lobulated and triangular lesion 40 mm  × 23 mm (transverse) × 16 mm (anteroposterior) in size in the anterior mediastinum, just anterior to the aortic arch  scan were normal. An echocardiogram is currently awaited.Following histology results, the patient discussed in the Urology MDT and subsequently referred to an oncologist, who suggested tumour markers namely beta human chorionic gonadotropin (βHCG), alpha-fetoprotein (αFP) and lactate dehydrogenase (LDH) levels which were negative and a repeat staging CT which was normal. The patient had a testicular ultrasound at the time of his original presentation, prior to his surgery, which excluded a testicular primary. A repeat positive emission tomography (PET) scan did not show any lesions either.Given the normal staging CTs, ultrasound, PET scan and negative tumour markers, the MDT decision was for active surveillance.Mediastinal tumours are most commonly thymomas, neurogenic tumours or benign cysts, altogether accounting for 60% of cases .. PrimarTumour markers such as βHCG and αFP are tumour markers which can be used diagnostically as well as follow up for recurrence. αFP is not secreted by primary mediastinal seminomas and therefore would either indicate a mixed germ cell tumour or testicular primary , 6. βHCGA review of the literature for primary mediastinal seminoma shows few recent case reports. The case series by Napieralska et al. looked at a single centers cases from 1983 to 2014. In this 31 year period they had 16 cases; 6 found incidentally after surgery as in our case . A case Also histologically, the background of florid follicular lymphoid hyperplasia, as seen in this particular case, is very rare. A paper by Weissferdt et al  looked aAt clinical follow up, 5 of the patients in the paper were alive and well 3 to 5 years after diagnosis; one patient was lost to follow up. The conclusion of this paper was that the true prognosis of these pattern of mediastinal seminoma with florid lymphoid hyperplasia needs to be investigated in a larger case series .At the time of writing of this paper, there were only three further publications that had reported similar cases. In the 2016, paper by Lee et al. had described a single case with the same immuohistological features, except for the presence of a synchronous thymoma . And finFirst line treatment in resectable mediastinal seminomas is surgical excision, with possible radiotherapy post-operatively depending on the status of histological margins , 6, 11.


Osteosarcoma epidemiology in children younger than 5-year-old is very rare. Currently, limb salvage surgery becomes the preferred treatment in osteosarcoma. Wide excision using cryosurgery has been reported as an effective and safe procedure for malignant bone tumors.A 5-year-old girl was presented with pain and a lump on her right knee. Physical examination showed a solid circumferential mass in the proximal tibia, with limited range of motion due to mass and pain. Osteoblastic lesion with a discrete margin and narrow transition zone on the proximal tibia from plain radiograph and magnetic resonance imaging (MRI) examination. Histopathological examination suggested osteosarcoma with a giant cell rich osteosarcoma subtype. Three cycles of neoadjuvant chemotherapy was conducted with cisplatin, ifosfamide, and adriamycin. We performed limb salvage surgery by wide excision with cryosurgery and vascularized fibular graft reconstruction. Wide excision was performed with the respect to preserve the epiphyseal plate. The proximal tibia segment was recycled using liquid nitrogen and re-implanted, fixed with a locking 2.7 mm T-plate and a straight reconstruction 2.7 mm reconstruction plate. Vascularized fibular graft was used to fill the bone defect on the medial side. Post-operative radiograph showed the plate and screws are well-fixated and the post-operative histopathological examination confirmed the diagnosis of conventional osteosarcoma HUVOS I. There was no post-operative complication observed, and the functional outcome was good.Cryosurgery and vascularized fibular graft is a viable reconstructive option for proximal tibia osteosarcoma in very young children. •Osteosarcoma in a very young child is rare.•Few cases of osteosarcoma in very young child treated with cryosurgery•Wide excision with cryosurgery and vascularized fibular graft reconstruction showed excellent functional outcome. The patient was with a history of falling from a bike two months prior to the admission with no significant injury related to the fall. General examination was within normal limit. The patient complains of unexplain loss of weight. Pain was felt out of proportion and worsen at night.Physical examination of the right knee showed a solid circumferential mass  on the proximal tibia . The ranThree cycles of neoadjuvant therapy regiment with cisplatin, ifosfamide, and adriamycin were administered to the patient. Multidisciplinary approach surgery with plastic surgeon and cardio thoracic vascular surgeon was conducted in this patient.The surgery was performed with a torniquet in a supine position with an anteromedial approach on the proximal tibia. The popliteal bundle was then identified and preserved. Ligation and anastomosis of the vein involved with the tumor were performed prior to wide excision. Wide excision was performed with the respect to preserve the epiphyseal plate proximally and 13 cm of the epiphyseal plate distally. The proximal tibia segment was clear from the tumor with osteotome and bone curette Fig. 5IntThe proximal tibia segment was recycled using liquid nitrogen for 40 min. The recycled autograft was left to be thawed at room temperature for 15 min and soaked in 160 mg gentamycin saline for 5 min . The recPostoperatively we performed an X-ray to evaluate the surgery. From the X-ray, the plate and screws are well-fixated . PostopePostoperative condition showed good surgical and functional outcomes. Postoperative complications did not occur in this patient. Radiological union was achieved in 1 year followup with the ROM of 120° in the affected knee. Functional outcome was calculated with musculoskeletal society tumor score (MSTS) with the result of 76,6%.3We presented the case of osteosarcoma in 5-year-old children treated with cryosurgery. Cryosurgery is the therapeutic use of cold to cause tissue necrosis with the goal of ablating the tissue. Because bone necrosis occurs at temperatures below −21 °C, cryosurgery with liquid nitrogen is beneficial in the treatment of bone malignancies. The major processes of liquid nitrogen induced cellular necrosis are thought to be the production of intracellular ice crystals and membrane rupture In this case, we performed a limb salvage surgery with adjuvant cryosurgery and vascularized fibular graft followed by hemigastrocnemius muscle flap. Limb salvage surgery is defined as a surgical procedure to preserve or restore the function of bone and joint after resection of a malignancy The use of liquid nitrogen as cryosurgery for osteosarcoma has been reported in a previous study [27]4Cryosurgery and vascularized fibular graft is a viable reconstructive option for proximal tibia osteosarcoma in very young children.None.None.Not commissioned, externally peer-reviewed.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Ethical approval was not required in the treatment of the patient in this report.Written informed consent was obtained from her parents and the patient gave assent 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.Sigit Daru Cahyadi contributes in the study concept or design, data collection, analysis and interpretation, oversight and leadership responsibility for the research activity planning and execution, including mentorship external to the core team.Uno Surgery Erwin contributes to the study concept or design, data collection and writing the paper.Does not need any registration.Sigit Daru Cahyadi, MD.The authors declare no conflicts of interest.