Epithelial downgrowth is a rare complication after Descemet stripping automated endothelial keratoplasty (DSAEK), which usually leads to poor visual outcome despite multiple available options of treatment.A 50-year-old man underwent DSAEK procedure due to pseudophakic bullous keratopathy. Three months later, the patient presented with gradual visual loss; slit-lamp examination revealed detachment and folding of the DSAEK lenticule, which was confirmed by anterior segment optical coherence tomography. On confocal scanning, epithelial cell sheets were detected in the interface leading to the wrinkling of the donor tissue and donor detachment. Surgical debridement and transient fixating with straight 10–0 prolene needles were performed followed by air injection into the anterior chamber. The cornea turned clear in the one-year follow-up with uncorrected-visual acuity of 20/30 and best-corrected visual acuity of 20/25.Early diagnosis and treatment of epithelial downgrowth may be associated with a good prognosis and prevent from more aggressive treatments such as repeat of grafting. In this case, mechanical debridement and transient fixation of lenticule by 10–0 prolene needles was performed to manage post-DSAEK epithelial downgrowth and lenticule detachment, which was successful without requiring of additional re-grafting. It seems this is a feasible technique with acceptable long-term outcomes.The online version contains supplementary material available at 10.1186/s12886-022-02264-7. Penetrating injuries and intraocular surgeries may be complicated with invasion of the epithelium into the anterior chamber, which is called epithelial downgrowth (ED) or ingrowth . MechaniTo the best of the author’s knowledge and database search, few cases of ED after DSAEK have been reported in the last decades. We report a case of lenticule detachment and epithelial downgrowth after Descemet Stripping Automated Endothelial Keratoplasty, which managed through surgical debridement and transient fixating of lenticule by straight 10–0 prolene needles not requiring a re-grafting.A 50-year-old man presented with pseudophakic bullous keratopathy after cataract surgery. His visual acuity was counting fingers at 1 m; the posterior capsule was intact with well-positioned intraocular lens (IOL) in the bag. The patient was scheduled for DSAEK surgery. In the DSAEK procedure, recipient corneal epithelium was removed and four venting incisions were made in the midperiphery of the cornea before Descemet stripping. The donor cornea was inserted using a Busin glide; after DSAEK lenticlue insertion, air injection was done up to the border of DSAEK lenticule. The surgical procedure resulted in a clear cornea. However, the patient presented with gradual visual loss three months later and the DSAEK lenticule was detached, non-mobile and folded confirmed by anterior segment optical coherence tomography (ASOCT)  , glaucoma filtering surgery, Descemet membrane endothelial keratoplasty (DMEK), and DSAEK may be complicated with ED . It seemPrevious studies have been shown that different risk factors such as several intraocular operations, poor wound closure, iris or vitreous incarceration, long-standing inflammation, corneal vascularization, and low intraocular pressure (IOP) may prone the eye to ED . MoreoveSubtypes of ED include epithelial sheets, pearls, and cysts. Sheet type is the most common type of ED as occurred in our patient. Also, this type is the most destructive type, which may cause vision loss through glaucoma and corneal endothelial failure . One stuThese patients usually turn symptomatic with decreased vision or pain. Non-specific features such as a hazy cornea, inflammation, and high IOP may make the diagnosis difficult. So, clinical suspicious can be helpful in these situations, especially in cases with history of previous ocular surgery. ED must be differentiated from iridocorneal endothelial (ICE) syndrome and metastatic carcinoma .The gold standard method of the diagnosis is a histological assessment, which can also differentiate between the corneal or conjunctival origin of ED through the presence of goblet cells in cases with conjunctival source. Also, argon laser photocoagulation has a diagnostic value, which shows whitening of burn spots on invaded iris surface whereas healthy areas appear dark. Other noninvasive diagnostic modalities include ASOCT, confocal scanning, and specular microscopy .Although there are some reports of spontaneous resolution of ED, various treatment options including surgical removal, cryotherapy, wide excision with adjunctive argon laser ablation, re-grafting through PK, DMEK, or DSAEK, and injection of antimetabolites like 5-FU into the anterior chamber are available. However, the failure rate of treatment is remarkable. It could be mentioned use of 5-FU was not successful in some cases . Informa2 was changed to 2109 cells/mm2.The unique aspect of our report is management of epithelial downgrowth without requirement to re-grafting. Unlike previous reports of epithelial downgrowth after DSAEK which led to re-grafting (re-DSAEK or PK) after a short duration despite primary management with debridement or using 5-FU, our patient is currently stable through our novel technique without re-grafting . It should be mentioned that the presence of epithelial sheets and diagnosis of ED was made using confocal scanning since unlike the typical cases of epithelial downgrowth, epithelial sheets were so fine in our complicated case leading to poor visibility through surgical microscope. It’s of note access and cleaning of epithelial sheets was done from venting incisions and interface scraping (but not excision) was tried to decrease the epithelial downgroth as less as possible. The authors have no claim regarding complete removal of epithelial sheets since this is not rationally possible and we think the reason of refractory nature of lenticule to repositioning is presence of remnant epithelial sheets. In this condition use of 10–0 prolene needles beside air injection was helpful in stretching of lenticule resulting in attachment. The concern of further endothelial damage by penetration of lenticule is logically right but it should be mentioned the needles have very tiny cross section and we believe endothelial damage following possible penetration was not significant. Our witness for this claim is comparison between preoperative and postoperative endothelial cell count (at 1st year of follow-up) in which 2327 cells/mmAt the time being, no study is available on direct comparison between outcomes of graft exchanging and debridement. Although this condition is so rare, future studies may be helpful to address the preferred option. Nevertheless, we deeply believe that giving a chance to the patients for prevention from re-grafting and its subsequent issues is a logical approach.In conclusion, early diagnosis and treatment of epithelial downgrowth may be associated with a good prognosis and prevent from more aggressive treatments such as repeat of grafting. In this case, mechanical debridement and transient fixation of lenticule by 10–0 prolene needles was performed to manage post-DSAEK epithelial downgrowth and lenticule detachment, which was successful without requiring of additional re-grafting. It seems this is a feasible technique with acceptable long-term outcomes.Additional file 1: Video 1. Shows the used surgical technique.


Malignant PEComa is a rare entity that usually origins at visceral, retroperitoneal and abdominopelvic sites. In the present paper, we describe an extremely unusual manifestation of malignant PEComa involving the mandible in a 48 years-old female patient focusing on the anatomopathological findings and differential diagnosis. The therapeutic management based on the head and neck tumor board indications is also discussed. Key words:Malignant PEComa, PEComa of the mandible, PEComa pathology, Oral cavity unusual neoplasm. PEComas (Perivascular Epithelioid Cells differentiation tumor) are a rare and anatomically ubiquitous family of mesenchymal neoplasms composed of nests and sheets of epithelioid cells with clear to granular eosinophilic cytoplasm and focal association with blood vessel walls . Its oriMost PEComas are benign. Malignant PEComas present an aggressive behavior, with potential for local recurrences, metastasis and death . The prePEComas presentation as a primary bone lesion is extremely rare. To our knowledge, only 11 cases of primary bone origin have been reported in the literature (excluding mandible). Pain is the most frequent clinical sign; swallowing and functional impotence are also common and a pathological fracture might happen if the osteolysis weakens the bone enough. Complementary image studies usually show an osteolytic lesion that associates a soft tissue mass and destruction of cortical bone in cases of high aggressivity .According to the therapeutic guidelines, surgical resection of primary tumor and its recurrences is the first option. Second therapeutic step involves adjuvant treatment with mTOR inhibitors, particularly in tumors with evidence of mTORC1 activation and somatic deletion of TSC1.We report the case of a 48-year-old female patient without relevant medical history, referred to our department  for the evaluation of a fast-growing and hyperemic surface soft tissue mass that involved the alveolar ridge of the left mandible. Specifically, the lesion had grown in the last two weeks. Pain and swallowing were the main symptoms; furthermore, the patient did not experiment inferior alveolar nerve paresthesia. The major diameter of the lesion was 4cm and mobility of dental pieces 37 and 38 was reached in association. The clinical evaluation of the neck was negative.An Orthopantomography (OPG) and a Computed Tomography (CT) scan were performed as diagnostic imaging techniques. The OPG evidenced erosion of the cortical bone subjacent to pieces 37 and 38 and the CT scan showed a tumor with smooth contour of approximately 27x40x47 cm involving left angle of mandible; not cervical dissemination was found Fig. .As first therapeutic approach, a conservative resection of the tumor was performed. The sample was analyzed by the Pathological Anatomy department of our institution in collaboration with the Pathological Anatomy department of the Hospital Universitario La Paz, Madrid. The anatomopathological analysis evidenced morphologic characteristics compatible with several entities, discarded based on different criteria. Conventional hematoxylin-eosin (HE) technique showed a neoplasm with diffuse growth pattern composed of epithelioid cells with an eosinophilic, clear and granular cytoplasm and inflammatory cells. The IHC showed positivity for histiocytic markers CD68, CD63, CD163 and cathepsin K. The muscle marker SMA positivity was quite focal. TFE3, MITF1 and the melanocytic marker KBA.62 turned out also positive. All the findings were compatible with PEComa  and the metastatic involvement of the neck, adjuvant treatment with Everolimus was administrate. Consequently, a partial response was achieved during seven months. However, when the adjuvant treatment ended, the patient suffered tumor persistence. Thus, palliative radiation therapy was administrated as a last therapeutic option, according to the consensus of the Head and Neck tumor board of our Hospital. Unfortunately, the patient died twenty months after the diagnosis as a cause of airway obstruction.The bone is rarely affected by PEComas and the mandible is an extremely unusual primary tumor location. To our knowledge, only one single case of malignant PEComa of the mandible has been reported previously in a 77 years-old female patient. The therapeutic management elected was the surgical resection. A segmental mandibulectomy was performed and the mandible was reconstructed with a prebent reconstruction plate. Adjuvant radiation therapy was posteriorly administered. However, MTOR inhibitors were not administered. The patient did not experiment any loco-regional recurrence up to 2 years after surgery .The differential diagnosis was complex due to the histological heterogeneity of the piece. The nuclearexpression of TFE3 has been described in a limited number of entities  that shaOther neoplasm with TFE3 as the epithelioid hemangioendothelioma was discarded due to the absence of distinctive morphologic characteristics and the negativity of CD34/CK .The histiocytic-like morphology of the cells and the expression of histiocytic markers have led to the wrong classification of the PEComas as tumors of histiocytic origin. However, lymphocytes plasma cells and or eosinophiles did not intermix in the lesion (as it happens in histiocytic sarcomas) and protein s100 and CD1a were negative (markers proper of Langerhans cells histiocytosis). Moreover, in the histiocytic lesions TFE3 positivity is not described . The maiIn regard to the therapeutic management, the surgical resection wasn´t effortless. The malignant behavior of the neoplasm and its fast growth led to an extensive locoregional infiltration, so the accomplishment of a R0 surgery was not possible. For this reason, the administration of MTOR inhibitors was justified as second therapeutic step ,15. Alth


Cervical thymic cysts are relatively rare benign cystic lesions that tend to be diagnosed clinically as branchial cysts, which usually present as painless, enlarging neck masses. They can occur anywhere along the normal path of descent of thymic primordia from the angle of the mandible to the sternal notch, with mediastinal extension observed in approximately 50% of cases. They are usually seen in the first decade of life on the left side with a male predominance. Here we report a case of a 15-year-old boy who presented to the hospital with left-sided neck swelling for about 2 months. The neck’s contrast-enhanced computed tomography (CECT) revealed a large, well-defined cystic swelling in the left neck region, showing peripheral enhancement, seen from the submandibular region to the superior mediastinum extending into the retrosternal region. Direct fine needle aspiration (FNA) was done, which showed a benign lesion with inflammatory and cystic characteristics, leading to the possibility of a branchial cyst. The cyst was completely excised surgically. Histopathology showed a thymic cyst with parathyroid tissue. The presence of thymic tissue with Hassall’s corpuscles is essential for the diagnosis. Knowledge of the clinical presentation, cyto-histological findings, and differential diagnosis of cystic cervical lesions in the pediatric population is important to diagnose this rare entity. Hence, though uncommon, when one comes across a cystic cervical region mass in children, a diagnosis of cervical thymic cyst should be kept in mind. Nonetheless, a definitive diagnosis depends on imaging findings as well as intraoperative findings and histopathological examination. Cystic neck swellings are usually related to developmental malformations. Anomalies of the lateral cervical region are distinctive to those of the midline anomalies. Cystic masses of the lateral cervical region occur due to developmental abnormalities involving the pharyngeal pouches as well as branchial clefts, which includes branchial cysts, lymphangiomas, and thymic cyst. In contrast, the midline anomalies occur due to abnormal closure of the midline or due to thyroid tissue remnants that persist after migration, which includes bronchogenic cysts, thyroglossal duct cysts and dermoid cysts.This case report aims to present the observation along with a review of literature and evidence on the clinical features, diagnostic aspects, and management.A 15-year-old boy presented to our hospital with a left-sided neck swelling for about 2 months. The patient noticed the neck swelling incidentally, which initially measured about 3x3 cm and progressed over 2 months to reach the present size of 11x4cm. There was no history of neck pain, fever, weight loss, loss of appetite, trauma, or difficulty in eating. On examination, the swelling was non-tender, non-pulsatile, non-translucent, mobile more in the vertical direction than horizontal direction with overlying normal skin temperature. The swelling did not move with deglutition or protrusion of the tongue. The neck contrast-enhanced computed tomography (CECT) revealed a large, cystic, well-defined swelling in the left neck region. The swelling showed peripheral enhancement and was seen from the submandibular region to the superior mediastinum extending into the retrosternal region .Direct FNA was performed using 23 gauge needle. 16ml of greenish-brown, clear to hazy fluid was aspirated, and the swelling apparently subsided after aspiration. The fluid was centrifuged, and post-centrifugation smears were subsequently stained with Papanicolaou, Hematoxylin and Eosin (H&E), and Leishman-Giemsa stains. Post-centrifugation smears were cellular and showed inflammatory infiltrate of cyst macrophages, lymphocytes, eosinophils, hemosiderophages, and occasional histiocytic giant cells. Few mature squamous epithelial cells, anucleate squames, proteinaceous debris, and numerous cholesterol crystals were noted in a fluidy proteinaceous background. No thyroid follicular epithelial cells were seen. The FNA was reported as a benign inflamed cystic lesion, and the possibility of an inflamed branchial cyst was given.Following the FNA diagnosis and imaging findings, the neck mass was resected A and wasAn intact brownish-grey cystic mass, 11.5cm in length, 80 grams in weight, was received with an elongated tubular projection at one end. The cystic mass measured 7x5x2.8cm, and the elongated tubular projection measured 4.5x2.2x2.6cm. Sectioning yielded about 14ml of turbid greenish fluid. The cut surface showed a large unilocular cyst with wall thickness ranging from 0.3 to 0.6 cm. The elongated tubular projection showed cyst with sieve-like areas B. HistopCervical thymic cysts are rare neck lesions with approximately less than 120 cases documented in the literature.,,,,The thymus originates from the third and fourth pharyngeal pouches and forms the main lymphoid organ during infancy and in children. The thymic primordia emanate from the pyriform sinus, passes through the lateral part of the thyroid gland and descends to the mediastinum. The thymic tissue can be found along this path of descent of the thymic primordia, due to which cervical thymic cysts can be found in the region from the mandible to the sternum.,,The thymic cysts are mostly elongated and range from 1cm to 26cm in diameter.A preoperative diagnosis is rarely formed, and the mass is removed by surgery which is the mainstay of treatment, which is both diagnostic as well as therapeutic. In 1901, Pollosson and PieryHsieh et al.The main entity to be differentiated is a branchial cyst that originates from the thymopharyngeal tract of the second pouch and lymphangioma. The patient's age and location of the cyst help in differentiating. The branchial cysts are located superficial and lateral to the internal jugular vein and common carotid artery, and ends in the superior tonsillar fossa and lymphangiomas are found in the posterior triangle of the neck. In contrast, thymic cysts pass posterior to carotid bifurcation, situated in close association with the carotid sheath and terminates in the pyriform fossa. Also, branchial cleft cysts rarely extend to the clavicle while thymic cysts tend to be larger, extending toward the anterosuperior mediastinum. Also, branchial cleft cysts are seen in young adults around the third decade of life, while thymic cysts tend to be pre-pubertal usually. Different imaging modalities are also helpful. Magnetic Resonance Imaging (MRI) is considered superior to ultrasonography (USG) and CT scan owing to its multiplanar capability and superior contrast resolution. On CT scan, the thymic cyst may appear as uni or multiloculated, hypoattenuating cystic mass present alongside the carotid space. No contrast enhancement is seen except in cases with solid components, which might represent aberrant thymic tissue, parathyroid tissue, or lymphoid tissue.,The differentials of cervical cystic neck masses are dermoid cyst, epidermoid cyst, thyroglossal duct cyst, laryngocele, hemangioma, bronchogenic cyst, thyroid lesions, cervical lymphadenopathy, rarely parathyroid cysts and malignancies.Cervical thymic cysts are rare but should be borne in mind in the differentials of cystic neck swellings, especially in the prepubertal age group. Definitive diagnosis depends on imaging modality, intraoperative surgical findings, and histopathological examination. A correct diagnosis can be rendered if the pathologists and clinicians are acquainted with this entity.


Tophacious gout presenting at the anterior cruciate ligament (ACL) is extremely rare and difficult to differentiate from other intraarticular pathology. This is mainly due to conventional diagnostic tools, such as MRI, producing ambiguous results versus pigmented villonodular synovitis (PVNS) and ganglion cysts.Here we report an individual in their late-20s with a gouty tophus located at the origin of the ACL in the knee. Urate crystals on the articular cartilage in all three compartments was noted as well as on the synovium. On advanced imaging with an MRI, a large mass was seen anteriorly in the notch surrounding the ACL and posterior cruciate ligament (PCL). The tophus was biopsied and excised arthroscopically with excellent results.An ACL mass in the knee has a very broad differential diagnosis. MRI imaging alone makes it very difficult to differentiate between PVNS and gout tophi yielding a pre-operative diagnostic challenge. Additionally, we review diagnostic challenges faced by other groups with similar cases, as well as their chosen treatment.Gouty tophi arising from the origin of the ACL are extremely rare and remain difficult to diagnose due to their ambiguous nature in conventional imaging. In this report, we clearly convey the disparity in the diagnostic protocol for this type of intraarticular pathology. Future research should look to develop a superior protocol for identifying these pathologies to improve diagnostic accuracy. •Atypical mass presenting at the origin of ACL, MRI unable to accurately diagnose.•Excisional biopsy revealed tophacious gout, 1 of only 4 cases presenting at ACL.•Multi-disciplinary approach needed in order to achieve a definitive diagnosis. Specifically, masses affecting or originating at the anterior cruciate ligament (ACL) are of particular interest. Common pathology include ganglion cysts, synovitis, pannus, gouty tophi and tenosynovial giant cell tumors, such as pigmented villonodular synovitis (PVNS). Ganglion cysts are masses with myxoid matrices most commonly occurring in connective tissue, although these are very rare . Tenosyn2A 29-year old male, formerly a competitive soccer player, presented with pain in the superior-lateral and posterior aspect of the left knee. He had a history significant for gout that was managed without medications. Physical examination revealed a moderate effusion and an extensor lag of 5 degrees. There was no laxity to the ACL or other ligaments. An initial MRI revealed soft tissue nodularity in the lateral and central aspects of the knee, most consistent with PVNS or gout , Fig. 2.3Intraarticular masses involving the ACL are rare . Here we, and not gout, which may explain the differing approaches [Treatment of ACL masses are fairly uniform throughout the reviewed literature. Most groups approached the mass arthroscopically and debrided to its base until successful excision ,10,11. Aproaches . If arthproaches . Continuproaches .4Gouty tophi arising from the origin of the ACL are extremely rare and remain difficult to diagnose due to their ambiguous nature in conventional imaging ,13. In oNot commissioned, externally peer-reviewed.None declared.This report is sanctioned by the Michael Garron Hospital REB Committee; ID: NR-303.Written informed consent was obtained from the patient for use of their case and medical imaging results. A copy of the written consent may be furnished upon the editors request.1.Name of the registry: Not applicable.2.Unique Identifying number or registration ID: Not applicable.3.Hyperlink to your specific registration (must be publicly accessible and will be checked): Not applicable.Jihad Abouali.EC: Drafting and revision. KR: Drafting, revision, approval of final manuscript. JA Drafting, revision, approval of final manuscript.None declared.


Formerly called dysembryoplastic neuroepithelial tumour (DNET) of the septum pellucidum, myxoid glioneuronal tumour (MGT) was recently recognized as a distinct entity. We report three cases of presumed MGT with typical location and image features. Besides, we present a literature review of the clinical profile, image findings and histological features of this new central nervous system tumour.A 59-year-old female came to the emergency department with complaints of chronic headaches. The neurological examination was unremarkable. The patient was referred to the radiology department for a non-contrast CT scan, which showed a hypodense, well-delimited lesion in the right septal area with no calcification or haemorrhage.1T weighted image (T1WI), hyperintensity on 2T weighted image (T2WI) and no gadolinium enhancement. On fluid attenuation inversion recovery (FLAIR) image, the lesion showed central hypointensity with a periphery rim of hyperintensity. Diffusion-weighted image (DWI) showed a hypointense signal with a corresponding high signal on the ADC map (facilitated diffusion) and a slight hypointense signal in the susceptibility-weighted image (SWI)  with hypointensity on ge (SWI) . Arteriage (SWI) .The patient is currently under conservative management in neurology clinics, and no surgical intervention was performed.A 49-year-old male was referred for a 3-month headaches history and nausea, associated with behavioural abnormalities, and two episodes of simple partial seizures. Brain MRI showed a well-defined lobulated lesion in the subcortical white matter of the right subcallosal gyrus and lamina terminalis, with the chiasmatic and infundibular involvement recesses and extension to the nucleus accumbens and anterior commissure.This lesion showed hyperintensity on T2WI, a slight hypointensity on SWI and no gadolinium enhancement. There was a central partially suppressed signal with a periphery rim of hyperintensity on the FLAIR image. There were no signs of restricted diffusion, calcification or haemorrhage . ASL, DSConsidering the clinical findings, this patient underwent a microsurgery resection without complications, and the diagnosis was septal DNET. The patient was discharged home the day after surgery. At a follow-up visit 3 months after surgery, the headaches had lessened in frequency. No more seizures were related.A 22-year-old male who underwent brain MRI was reporting a 7-month history of disconnected speech and upper limb movements, along with sporadic episodes of loss of consciousness lasting approximately 15 to 20 s. These episodes increased gradually as frequently as twice a day.Brain MRI showed a sizeable well-delimited lesion with an “L” shape in the left septal area, inferior aspect of the septum pellucidum, with extension into the third ventricle . This leThe patient underwent open craniotomy with partial resection. The pathological diagnosis was septal DNET. No follow-up scan was performed until this publication.2 Besides the PDGFRA mutation, no additional pathogenic mutations were identified.1,2 The cIMPACT6-Utrecht meeting suggested that MGT should be considered a distinct tumour type and could be placed within the section covering “Neuronal and mixed neuronal-glial tumours” as WHO Grade I lesion.The MGT was first described as a single entity in 2018 by Solomon et al that demonstrated an isolated mutation in the PDGFRA gene resulting in dinucleotide substitution at codon p.K38.5 The majority can be cured by surgical excision, and a minority needs radiation or chemotherapy. With few exceptions at histology, these tumours are classified as Grade I, with morphologically variable and usually occurs in children and young adults. Headache is the most common symptom, followed by seizures, emesis, behaviour disorders, visual disturbances, altered mental status, hydrocephalus, facial drop and personality changes.1,2,6 Some patients can be asymptomatic, and the tumour is incidentally discovered in brain imaging studies.These tumours are characterized by the presence of neoplastic ganglion cells and glial  elements and represent up to 2% of all CNS tumours.All three cases had a presumptive diagnosis of MGT based on the typical location and MRI features. As demonstrated by Salomon et al and suggested by cIMPACT6-Utrecht meeting, the MGT characteristically involves the septum pellucidum, septal area, subcallosal area, corpus callosum and lateral ventricle. CT does not play a significant role in TGM characterization. It frequently shows a hypodense lesion with a pseudocyst appearance, but no true cysts are seen histologically. There was no calcification or acute haemorrhage.7,8 The 3D highly T2WI sequence  with high liquid-brain contrast demonstrates a slightly decreased signal compared with the cerebrospinal fluid, confirming that it is a solid lesion. Diffusion-weighted image shows facilitated diffusion and helps differentiate it from the multinodular vacuolating neuronal tumour (MVNT), that was recently described as a true neoplastic process.9,10 TGM shows high ADC values compared with normal brain parenchyma because of their low cellularity. Lack of oedema is characteristic. Large septal lesions may show a distinct “L”-shaped appearance and can do a critical mass effect, suggesting another high-grade neuroepitelial tumour; his specific location is an excellent guide for the diagnostic hypothesis.11 However, often due to the compressive effect and psychological symptoms, surgery is necessary.The conventional MRI clarifies the findings and usually shows a well-defined lobulated mass, with hypointensity on T1WI, hyperintensity on T2WI, and no gadolinium enhancement. FLAIR imaging is essential to this new entity’s diagnosis, as it shows a partially suppressed signal in the lesion center, with a peripheral rim of hyperintensity, classically described in the DNET.12 They are labelled by inverting their spin polarity (preparation phase) and a delayed time when perfusion-weighted images are acquired contrast use. ASL in the Neuronal and mixed neuronal-glial tumours do not demonstrate high CBF like in high-grade gliomas. These values are an essential non-invasive and fast parameter that helps in grading and differentiating the central nervous system tumours.12Another recent MRI method used to evaluate blood flow using electromagnetically labelled arterial blood water is arterial spin labeling. This technique uses radiofrequency pulses to magnetize the inflowing blood water protons.13,14 The cysts are typically attached to the anterosuperior portion of the third ventricular roof. They rarely are found at other sites, including the lateral ventricles, cerebellar parenchyma, pituitary and various extra-axial locations.14,15The most important differential diagnosis of MGT located in the septum pellucidum is the third ventricle’s colloid cyst. The colloid cyst usually presents a hyperattenuating mass on CT, high signal intensity on T1WI, and variable signal intensity on T2WI in MRI., attached to lateral ventricles' septum pellucidum walls. Calcification is common (50%), as likely as variable and heterogeneous post-gadolinium enhancement.6,16 Subependymoma is a rare slow-growing tumour fourth ventricle in middle-aged to elderly patients, and when it protrudes into the lateral ventricles, more often than into the third ventricle. It usually does not enhance on post-contrast sequences; however, it is a solid glial lesion with a central hyperintense signal on the FLAIR image.17 Beyond these lesions, it is essential for a radiologist to know the normal anatomy and exclude a pitfall by CSF flow artefact simulating lesion.15Central neurocytoma and subependymoma are tumours that can also occur near the septum pellucidum. Central neurocytoma characteristically demonstrates a solid-cystic appearance, well-circumscribed mass1,2,7 Recent studies found that immunohistochemistry neuronal components are negative to FGFR1 mutations or rearrangements, which characterize most of the cortical DNTs or rosette-forming glioneuronal tumours negativity to BRAF, RAF1 or FGFR1 mutations or fusions present in most pilocytic astrocytomas or other low-grade neuroepithelial tumour entities.1,2,5 The multi-nodularity typical pattern of cortically based DNET is also absent.Histologically, MGT demonstrates a low-grade proliferation of oligodendrocyte-like tumour cells embedded in a prominent myxoid/mucin-rich stroma. Additionally, a fine capillary network reminiscent of DNET or oligodendroglioma is present. In some cases, it may demonstrate neurocytic rosettes.1,2 This behaviour is essential to make the correct diagnosis of MGT and its differentiation from other benign lesions, like colloid cysts, subependymomas or MVNTs.Despite being classified as a WHO Grade I tumour with relatively benign behaviour, intraventricular dissemination and local recurrence have been described.Myxoid glioneuronal tumour is a new entity with a distinct isolated genetic mutation recently described. For that, in the cIMPACT6-Utrecht meeting, this lesion was considered a distinct tumour type. It could be placed within the section covering “Neuronal and mixed neuronal-glial tumours” as WHO Grade I lesion.The interpreting radiologist should recognize the MGT’s imaging features, mainly of this specific location – septal area, corpus callosum and lateral ventricles – and this relatively benign behaviour evolution.MGT’s imaging features should help differentiate him from other lesions in this area, like a colloid cyst, central neurocytoma or subependimoma.


Retropubic parasymphyseal cysta are rare, and few cases have been reported in men.A 65‐year‐old male patient presented with a 6‐month history of pelvic and perineal pain. Magnetic resonance imaging revealed a high‐intensity, irregular‐shaped mass extending from the pubic symphysis to the bladder. Contrast enhancement revealed no uptake in the central part of the mass, indicating a cystic component. Computed tomography showed erosion of the pubic symphysis and pubic osteophytes. Pathological findings of biopsy specimens revealed inflammatory fibrous tissue but no malignancy. The definitive diagnosis was retropubic parasymphyseal cyst associated with inflammation. The patient was treated with cefazolin from 1 day before surgery until postsurgical day 7. Oral antibiotic therapy was then prescribed for 1 month to maximize treatment. After 2 months, the patient’s symptoms resolved.Retropubic parasymphyseal cysts with inflammation and smaller asymptomatic cysts can be managed effectively with conservative or minimally invasive treatment. ALPalkaline phosphataseCRPC‐reactive proteinCTcomputed tomographyMRImagnetic resonance imagingRetropubic parasymphyseal cyst is a rare lesion that can cause severe symptoms. Careful evaluation is warranted to differentiate these lesions from malignant tumors and abscesses. A multidisciplinary team, including urologists, pathologists, and radiologists, is crucial for diagnosing and treating atypical cases such as this one. Retropubic parasymphyseal cysts with inflammation, as well as smaller asymptomatic cysts, may be managed effectively with conservative or minimally invasive treatment. After 2 months of antibiotic treatment, our patient’s symptoms resolved, and serum C‐reactive protein and alkaline phosphatase levels were normalized.Retropubic parasymphyseal cysts are rare, and few cases have been reported in men. Retropubic parasymphyseal cysts may be asymptomatic or cause symptoms, such as urinary tract manifestations and pelvic pain. Because of the location, these cysts may be confused with malignant tumors and abscesses. In women, these lesions are thought to be caused by postmenopausal reactive changes as a result of multiparity.A 65‐year‐old man presented with a 6‐month history of lower abdominal, pelvic, and perineal pain. His medical history was unremarkable. He had previously been evaluated for prostate disease by a urologist, but no abnormalities were found.At admission, his urinalysis yielded normal findings; however, ultrasonography revealed thickening of the bladder wall. Cystoscopy demonstrated edematous regions in the anterior wall of the bladder Fig. . SagittaAfter case review, the multidisciplinary team recommended drainage of the atypical cyst and pathological examination to rule out malignancy. The pelvic pain hindered the patient’s mobility; thus, prompt diagnosis and treatment were necessary. He therefore underwent exploratory laparotomy.We reached the pelvic cavity in laparoscopic procedure. No abscess was observed in the pelvic cavity; however, a cyst with inflammatory, hard fibrous tissue was observed around the pubic symphysis. We collected tissue specimens using forceps and placed a drainage tube over the cyst as laparoscopic fenestration. Only a small amount of serous drainage flowed out, and the drainage tube was removed after a few days.A biopsy sample of the lesion was obtained, and pathological findings revealed inflammatory fibrous tissue with lymphocytes but no malignancy Fig. . The culThe patient was treated with cefazolin sodium 1 g IV q8h starting 1 day before surgery and continuing to postsurgical day 7. The patient’s symptoms rapidly improved, with serum CRP and ALP levels improving significantly. We continued antibiotic therapy minocycline 100 mg PO q12h for 1 month for optimal results. Over the next 2 months, the patient’s symptoms resolved, and serum CRP and ALP levels normalized.Retropubic parasymphyseal cysts, are rare lesions that develop in the pubic symphysis, were first reported in 1996.In a literature review of 18 patients with retropubic parasymphyseal cysts, including 2 men and 16 multiparous women, Taniguchi et al.In multiparous women, retropubic parasymphyseal cysts are thought to result from postmenopausal reactive changes.We did not perform needle biopsy because the mass could not be identified by ultrasonography, and CT‐guided aspiration from an anatomically difficult location may cause tumor contamination, as reported previously.Exploratory laparotomy enabled fenestration of the mass, and antibiotic treatment was effective, as evident from the lack of symptoms at the 2‐month follow‐up. Communication among the multidisciplinary team, which included urologists, pathologists, and radiologists, was crucial for diagnosis and treatment in this atypical case.In rare retropubic parasymphyseal cyst cases, invasive surgery might be necessary when symptoms are severe, but conservative treatment or minimally invasive therapy may be effective and should be considered, as in this case report.The authors declare no conflict of interest.Not applicable.We obtained consent from our patient for the publication of this case.Not applicable.


Paragangliomas arise from neural cells and are found in different anatomical locations in the body. Paragangliomas in the adrenal glands are called pheochromocytoma, while the others are known as extra-adrenal paragangliomas. They are usually benign and are extremely rare in children. We present a case of a 13-year-old female patient who presented with complaints of hematuria for one year and left lower lumbar pain. Imaging investigations depicted a urinary bladder mass that was causing a mass effect at the left ureteric opening and backpressure changes in the left kidney. The patient underwent transurethral resection of bladder mass, and the histopathology confirmed the presence of paraganglioma. Though the paragangliomas of the urinary bladder are extremely rare in the pediatric age group, we suggest keeping paragangliomas on differentials when investigating a patient with bladder mass. Pheochromocytoma and paraganglioma are neural cell origin tumors . Being hParagangliomas in the urinary bladder being a rare entity has a prevalence of around 0.06% of all tumors, and those with malignant potential advocated by metastasis are extremely rare ,4. The fA 13-year-old girl presented to the emergency department with a history of hematuria for the last one year associated with on and off left lumbar pain. Her family history was insignificant for any malignancy of the genitourinary tract. Her pulse was 94 beats/minute, blood pressure was 150/90 mmHg, and respiratory rate was 17 breaths/minute. Here general physical and abdominal examination was unremarkable.Ultrasound (USG) of kidneys and urinary bladder was performed with a full bladder, and an irregular mass was appreciated in the left posterior wall of the urinary bladder with significant left-sided hydronephrosis Figure .Contrast-enhanced computed tomography (CT) confirmed a large urinary bladder mass in the left margin of the urinary bladder base. It had heterogeneously enhancing soft tissue density and was abutting the anterior perirectal wall and the adjacent structures. Multiple enlarged lymph nodes were seen in the left pelvic area Figure .The multidisciplinary team decided to proceed with transurethral cystoscopic resection of the mass with special preoperative and intra-operative preparation (phenoxybenzamine and propranolol) to prevent any hypertensive crisis. Surgery and postoperative recovery went uneventful. Histopathology of the resected specimen revealed a focus of tumor cells within the bladder mucosa. The tumor cells were arranged in nests and trabeculae. Individual cells were round to oval with abundant cytoplasm and atypical nuclei. The immunohistochemistry was significant for chromogranin, synaptophysin, and S100 positivity Figure .A diagnosis of urinary bladder paraganglioma was made. The patient was discharged on the third postoperative day and is living a routine healthy life to date.Paragangliomas originating in the urinary bladder are extremely uncommon tumors that grow from chromaffin tissue of the sympathetic nervous system related to the urinary bladder. These growths can be active (producing secretions) or inactive (not producing any secretions) . The parParagangliomas occurring in the urinary bladder make 0.05% of all tumors. In the genitourinary system, the urinary bladder is the most frequent site for the occurrence of paragangliomas 79.2%), and the second most common site is the urethra (12.7%) followed by the pelvis (4.9%) and ureter (3.2%). Female predominance has been reported in the literature with a female-to-male ratio of 3:1, and they usually present between 30 and 50 years of age . Most tu9.2%, andDue to the absence of typical features, these are sometimes misdiagnosed as urothelial cancers. Around 10% of the paragangliomas in the urinary bladder are malignant . Gross oThe paragangliomas of the urinary bladder usually have a lobulated macroscopic appearance. On microscopy, the tumor cells are arranged in trabeculae and nests making the zellballen pattern. Cells have acidophilic granular cytoplasm with oval-shaped nuclei . AlthougParagangliomas can occur in a variety of anatomical locations.. The imaging features of paragangliomas of different locations are almost identical, and local or distal metastasis differentiates between benign and malignant lesions. We suggest keeping paraganglioma in the differentials of a solid urinary bladder mass lesion in pediatric age group patients.


Staphylococcus aureus. Hindlimb ataxia was attributed to infectious sacroiliitis and its secondary retroperitoneal abscess. As far as the authors' knowledge, this is the first report of multimodality imaging of infectious sacroiliitis with retroperitoneal abscess caused by S. aureus in a dog.A 3-month-old intact male Labrador Retriever was presented for falling trauma and hindlimb ataxia. Radiography indicated radiolucent left sacroiliac joint with irregular margin. Computed tomography revealed thickened sublumbar muscles and hypoattenuated sacroiliac joint while magnetic resonance imaging demonstrated abscess at retroperitoneum and gluteal muscle. Ultrasonography showed lytic left sacroiliac joint with retroperitoneal fluid, and fine needle aspiration resulted  Pasteurella and Brucella have been reported as causative agents in veterinary literatures, but Staphylococcus aureus is the most common agent in humans  and high C-reactive protein level . Along with toxic changes of leukocytes, many juvenile leukocytes and band cells were noted in the blood smear examination.Thoracic radiography  was unremarkable. However, abdominal radiography revealed irregular ventral margin of 4th lumbar vertebra and decreased abdomen serosal detail . Young aAbdominal ultrasound performed  with linear-array (10 MHz) probe revealed bilateral sublumbar and inguinal lymphadenopathy . ModeratTo further characterize the bone lysis of sacroiliac joints and the irregular margin of the lumbar 4th vertebra, computed tomography (CT) examination was performed . Imaging parameters were slice thickness 2 mm, pitch 0.8, tube rotation time 0.5 s, 130 kVp, 100 mAs, 512 × 512 matrix. Left sacral bone and ilium exhibited lower bone attenuation compared to the right sacral and ilium . Left su*) images in transverse plane . Slice thickness was 3–4 mm and intervals were 0.3–0.85 mm. Post-contrast T1W images were acquired after administrating 0.15 mmol/kg of gadolinium-based contrast agent intravenously . No remarkable changes were noted in lumbar spinal cord on T2W images (Magnetic resonance imaging (MRI) examination of the lumbar spinal cord was performed with 1.5 T scanner . The study included T2-weighted (T2W) images in sagittal and transverse planes , pre- and postcontrast T1-weighted (T1W) images in sagittal and transverse planes , and gradient echo  probe was performed to evaluate the bone margins of the sacroiliac joints and soft tissue changes. Bone lysis of the left sacrum and the iliac wing was noted . AnechoiStaphylococcus aureus and cefazolin to be effective against the microorganism. After antimicrobial treatment, the patient showed improved appetite and hindlimb condition. The patient still showed some hindlimb ataxia but was able to support his body weight with his legs and ambulate. Due to the owner's request, the patient was discharged. At 1 week follow-up after the diagnosis, the patient started rehabilitation therapy along with antimicrobial treatment. Prognosis of the patient was regarded as very good because the patient's condition improved dramatically 3 days after the antibiotic treatment and was able to ambulate 1 week after. At 1 month follow-up after the diagnosis, the patient did not exhibit hindlimb ataxia.Considering the findings of the imaging studies and the cytologic evaluation, infectious sacroiliitis, myositis, and retroperitoneal abscess were likely. To prevent sepsis, antibiotic therapy was warranted. While pending for the antibiotic susceptibility test result, the patient started with cefazolin , famotidine , and fluid therapy , which dramatically reduced leukocytosis and C-reactive protein level. Antimicrobial analysis indicated This report presents hindlimb ataxia and neurologic deficits in patients with retroperitoneal abscess and infectious sacroiliitis. The abscess in the retroperitoneal space was secondary to sacroiliitis along with retroperitonitis. Also, infectious sacroiliitis caused gluteal and sublumbar muscle myositis. These findings could have all contributed to hindlimb ataxia.Pasteurella canis and Brucella canis have been reported as causative agents  or fat-suppressed sequence of MRI demonstrating abscess is another limitation of this study. Nonetheless, recognition of fluid in the retroperitoneal space and successful FNA under ultrasonography guidance led authors to correctly identify retroperitoneal abscess.In some private practices, not all imaging modalities mentioned in this study are available. All four modalities  contribute to the diagnosis but some could be unnecessary. Radiography and ultrasonography are necessary to visualize the bone lysis and adjacent retroperitoneal changes. MRI is also indispensable to rule in/out spinal cord diseases. However, CT could be unnecessary since bone lysis could be depicted in radiography and retroperitoneal abscess in ultrasonography or MRI.S. aureus. Ultrasound-guided FNA of sacroiliac joint is a powerful technique that allows correct diagnosis of infectious origin and is strongly recommended.In conclusion, infectious sacroiliitis and retroperitoneal abscess should be considered when a young dog with hindlimb ataxia and neurologic abnormalities exhibits lytic sacroiliac joint with retroperitoneal fluid with adjacent inflammation. To the authors' knowledge, this is the first report to demonstrate multimodality imaging of infectious sacroiliitis and retroperitoneal abscess caused by The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.Ethical review and approval were not required for the animal study because this was a retrospective case report. Written informed consent was obtained from the owner for the publication.JJ and HY contributed to manuscript writing and image interpretation. HY contributed to radiography, ultrasonography, CT, and MRI scanning. JKo, JKi, and KE assisted image interpretation. YC, KL, JJ, and HY contributed to manuscript editing. All authors reviewed and approved the final submitted manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


A diaphragmatic hernia is a protrusion of abdominal contents into the thoracic cavity. Although it is commonly congenital, diaphragmatic hernias can also be acquired. Blunt or penetrating trauma are among the most frequent causes, although spontaneous or iatrogenic cases have been reported. Recently, some case reports related to diaphragmatic hernia after debulking surgery for advanced ovarian cancer have been described. This is an exceedingly rare but life-threatening complication, being prompt recognition and surgical correction critical.We report a case of a delayed diaphragmatic hernia in a 19-year-old female resulting from cytoreductive surgery for advanced ovarian cancer. Rapid evolution from gastrointestinal symptoms to hypovolemic shock occurred, and intensive care admission was required. Immediate surgery was critical to improving the patient outcome.This case highlights this uncommon but life-threatening complication, the challenges of diagnosing and managing those patients, and the need for early recognition, support, and surgical correction. Peritoneal invasion is a common feature in patients with advanced ovarian cancer, and at least two-thirds of patients with this type of cancer present with FIGO  stage III or IV disease at the time of diagnosis .It seems that the foremost important prognostic indicator in patients with advanced-stage ovarian cancer is the volume of residual disease after surgical debulking ; therefore, a surgical procedure with extensive cytoreduction is advised . PatientThe most frequent complications associated with diaphragmatic peritonectomy comprise pleural effusion, pneumothorax, and pneumonia . HoweverWe report a case of a diaphragmatic hernia occurring three months after cytoreductive surgery for advanced ovarian cancer.A previously healthy 19-year-old female was diagnosed with serous ovarian carcinoma, complicated with peritoneal carcinomatosis, in July 2019. By the time of August 2019, the patient underwent complete peritonectomy with a total hysterectomy and bilateral salpingo-oophorectomy. The postoperative course was uneventful. She received three courses of adjuvant chemotherapy with carboplatin over the following three months as consolidation therapy.She was admitted to the emergency room in November 2019 with gastrointestinal complaints, including abdominal pain, vomiting, and watery diarrhea, lasting 24 hours until the admission. On admission, her physical examination was remarkable for afebrile, dehydration, hypotension (blood pressure 67/32 mmHg), and sinus tachycardia (pulse rate 120 beats/minute). An enlarged abdomen accompanied by tenderness was noted. Laboratory findings revealed elevated hemoglobin (18.6 g/dL), although with normal white blood cell and platelets count; serum urea was 73 mg/dL, creatinine 1.81 mg/dL, and this was accompanied by hypokalemia (2.96 mmol/L), normal sodium and chloride concentration, 141 mmol/L and 99 mmol/L, respectively. A C-reactive protein concentration of 2.31 mg/dL was measured; liver enzymes, total bilirubin, and lactate dehydrogenase were within the normal range. Arterial blood gas revealed metabolic acidosis and extreme hyperlactacidemia (8.95 mmol/L).A standard X-ray of the abdomen was performed, showing dilatation of both small bowel and colon with scarce air-fluid levels; a standard chest X-ray was also performed, and a minor diaphragmatic defect was suspected Figure . A compuTaking into account the clinical and radiological data, an upper endoscopy was urgently performed, showing signs of gastric mucosal ischemia Figure . The herThe patient presented with fever on the third postoperative day, and the chest standard X-ray showed a pleural effusion. A diagnostic thoracentesis was performed, unveiling an empyema. She was started on empirical IV antibiotics, and a pleural catheter drain was inserted. The remaining postoperative course was uneventful, with complete clinical and radiological recovery Figure . She wasThe extensive upper abdominal surgical procedure, including diaphragmatic peritonectomy, is highly advised for maximal cytoreduction in patients with advanced ovarian cancer exhibiting peritoneal carcinomatosis. Diaphragmatic peritonectomy improves patient outcomes, and it is associated with a better prognosis , yet it A delayed diaphragmatic hernia is a rare complication after cytoreduction surgery, and very few cases have been reported in the literature -8. Left-Some of the reported cases of iatrogenic diaphragmatic hernias after debulking surgery have also been associated with hypothermic intraperitoneal chemotherapy (HIPEC) -8; it waClinical presentation of diaphragmatic hernia can vary from mild and nonspecific symptoms, such as vomiting and abdominal pain, to severe gastrointestinal and respiratory symptoms with hemodynamic instability. As a result, this complication is usually misdiagnosed, and sometimes the diagnosis is overlooked until strangulation of the hernial content occurs . In mostComputed tomography is considered the gold standard technique to establish the diagnosis, allowing the evaluation of the size, location, and content of the diaphragmatic defect . DespiteThe treatment of choice for diaphragmatic hernia is surgery. Patients with a symptomatic hernia should undergo urgent surgery, while in case of asymptomatic diaphragmatic hernia the repair procedure may be elective. Regardless of the size of the defect, all diagnosed diaphragmatic hernias require surgical repair to avoid serious complications such as incarceration, strangulation, and rupture . In compWe report a case of a delayed diaphragmatic hernia after cytoreductive surgery with diaphragmatic peritonectomy for advanced ovarian cancer. This is a rare complication, challenging to predict and diagnose. Reviewing the patient’s surgical history and recognizing the symptoms are crucial to early detection of this potentially life-threatening complication. Surgical repair is the treatment of choice for iatrogenic diaphragmatic hernia.


Additional anatomical structures are rare but can be mistaken for other conditions, causing misdiagnoses and poor outcomes for patients. The presence of concurrent anomalies within the extra structures further complicates a rare situation. We present a case of a patient with two gallbladders and a choledochal cyst diagnosed via radiography and confirmed by exploratory laparotomy. He underwent a cholecystectomy, choledochal cyst resection, and hepaticojejunostomy, and he was doing well as of his last follow-up. This case highlights the need to consider radiological imaging in patients with choledochal cysts carefully. Knowledge of anatomy and a high level of alertness for congenital abnormalities is critical for safe surgical operations. A double gallbladder is a rare anomaly of biliary disease, with an incidence rate of one in 3,000 to 4,000 cases . A doublA 26-year-old man presented to our clinic with recurrent right upper quadrant abdominal discomfort lasting one month accompanied by feelings of nausea without emesis. He had no yellowish discoloration of the skin and no abdominal swelling or fever on physical examination. The patient stated he was not constipated, nor does he have familial history of malignancy. The results of his laboratory workup, including complete blood count and liver function tests, were within reference ranges. An abdominal ultrasound revealed duplicate gallbladders. Magnetic resonance cholangiopancreatography (MRCP) confirmed the presence of a second gallbladder and a type I choledochal cyst Figure .We conducted an exploratory laparotomy which confirmed the radiology findings. We began the dissection by mobilizing the head of the pancreas. Blunt dissection and electrocautery were used to open the hepatic portal system, while the common bile duct was given a wide margin to avoid injuring the portal vein. We cleared the lateral, posterior, and medial attachments and mobilized the two gallbladders using electrocautery.We mobilized the pancreas entirely using blunt and sharp dissection Figure .The large vessels were double clamped with the distal intrapancreatic joint bile tube. We tied and divided the distal joint bile duct 0.75 cm beneath the right and left hepatic tubes Figure .We created a jejunostomy after identifying a loop of jejunum, and we created a mesocolon window then conducted a hepaticojejunostomy. We placed a drain after confirming there were no leakages from the anastomosis. We sent samples for histopathological examinations, which revealed a choledochal cyst and a double non-communicating lumen lined by muscularis propria, columnar epithelium, and lamina propria adjoined to the adventitial layer. The patient recovered from the surgery with no complications and was doing well at his one-year postoperative follow-up.Double gallbladder concurrent with a choledochal cyst is uncommon. Boyden classified the gallbladder’s association to the cystic duct into two main classes: split-primordium (doubled) or accessory gallbladder . HarlaftCholedochal cysts are categorized according to the degree of biliary tract dilation and location . Type 1 The clinical presentation of extra gallbladder concurrent with choledochal cysts can range from no symptoms to intermittent abdominal pain, jaundice, fever, or even cholangiocarcinoma. Double gallbladder does not present with any specific symptoms, does not increase disease possibility in either lobe, and is usually of little clinical significance unless disease is present. Diagnostic modalities start with obtaining transabdominal ultrasonography, given its high sensitivity for diagnosing gallstones, cholecystitis, or other anomalies. However, since double gallbladder can mimic other pathologies, further investigations may be required, including MRCP, computed tomography, cholangiography, percutaneous transhepatic cholangiography, or intraoperative cholangiogram to delineate the duct structures.There is no role for prophylactic cholecystectomy in accidentally diagnosed double gallbladder. However, when it is symptomatic, the management of double gallbladder can be done laparoscopically or via open surgery .This case represents the first report of two relatively rare congenital conditions  in the same patient. Double gallbladder is an unusual congenital condition that may mimic a choledochal cyst, even on an investigation such as MCRP. Surgeons should differentiate between the two abnormalities and remove the gallbladders when the patient is symptomatic. Later, they can perform an open or laparoscopic procedure, depending on the operating surgeon’s expertise. Knowledge of anatomy and a high level of alertness for congenital abnormalities is vital for safe surgical operation and optimal patient outcomes.


Echinococcus multilocularis. It nearly always occurs in the liver, and cardiac involvement is extremely rare. Liver resection is the most effective intervention for AE because the only potentially curative treatment is removal of the lesion. Even when complete resection is not performed, long-term survival can be expected after surgical removal of most of the lesion with lifelong administration of albendazole (ABZ).Alveolar echinococcosis (AE) is a rare parasitic disease caused by the larva of A 64-year-old man who lived in Hokkaido was referred to our hospital due to abnormalities in biliary enzymes. According to the findings from enhanced computed tomography and magnetic resource imaging of the abdomen, transthoracic echocardiography and serologic tests, he was diagnosed with hepatic AE with rupture into the pericardium. He underwent extended left hemi-hepatectomy with reconstruction of the inferior vena cava and opening of the pericardium with drainage as reduction surgery. Pathological examination revealed echinococcal infection in the pericardium as well as the liver. He started chemotherapy with 400 mg ABZ per Day 67 days after surgery. Although the surgical margin was positive in the pathological findings, he was alive 19 months later with no regrowth of the echinococcal lesion.AE with cardiac involvement is extremely rare. Even if the complete removal of cardiac-involved AE is not possible, surgical debulking with lifelong ABZ treatment can successfully manage the disease. Echinococcus multilocularis. This parasite introduces various patterns of hepatic lesions, and it is somewhat different from echinococcosis caused by Echinococcus granulosus, a hydatid species that is common worldwide .–14.11–14The first case of cardiac alveolar echinococcosis was recently published . The patThis is the first report of hepatic AE with cardiac involvement treated by the combination of surgery and ABZ. Even in cases like this, a surgical procedure with postoperative ABZ can be a useful treatment.This is the first report of (extremely rare) hepatic AE with cardiac involvement that was treated with surgery and ABZ. Even in the case of cardiac involvement, surgery followed by ABZ can be a promising treatment.


Endometrial stromal sarcoma is a rare tumour. It is even rarer to find it arising in the background of endometriosis in an extrauterine location. This case report describes a case of missed diagnosis of intestinal extra-uterine endometrial stromal sarcoma associated with endometriosis, and the subsequent presentation with distant metastases. The potential pitfalls are highlighted and differential diagnoses are discussed. Endometriosis is quite common in the reproductive age group with a reported incidence of 10% ,3. MaligESS is characterized by cells that resemble proliferative phase endometrial stromal cells. It usually originates from the uterine corpus, but may be seen in extra-uterine locations . ESS ariWe report a case of missed diagnosis of intestinal extra-uterine endometrial sarcoma associated with endometriosis, and subsequent presentation with distant metastases.A 46-year-old female patient was admitted to the hospital with abdominal pain and vomiting and had a CT scan of the pelvis which showed a complex adnexal mass and large mixed density mesenteric lesion in association with small bowel loops (20 x 15 cm). The patient underwent laparotomy where the mesenteric tumour and small bowel loop were excised and sent for histological examination along with a clinical diagnosis of possible mesenteric lymphangioma or endometriotic cyst. There was no definite adnexal mass found during surgery and liver and rest of the bowel were unremarkable.The histological examination of the mesenteric mass showed benign-looking endometrial type glands surrounded by monotonous endometrial stromal cells which revealed no obvious cytological atypia Figure . ImmunohThe patient recovered well after surgery and was discharged in a satisfactory condition. However, after 14 months from the first admission, she presented to the accident and emergency department (A&E) with abdominal pain.CT scan of the chest, abdomen, and pelvis was done which showed cystic solid liver lesions, pulmonary micronodules, omental infiltration, and soft tissue deposits. A suspicion of metastatic malignancy was raised at this point and correlation with the previous histology examination of the mesenteric mass was advised.An ultrasound-guided liver biopsy was performed and sent for histology. The liver biopsy showed complete replacement by spindle cells with a positive expression for CD10 and ER Figure , insets.It was noted that the changes were of similar appearance to the stromal changes in the small bowel resection received earlier. A possible diagnosis of a stromal component in endometriosis or ESS was considered. After external review, it was agreed that the liver biopsy contained ESS and the appearances were in keeping with those of metastatic ESS in which the tumour is of uniform “low grade” appearance.This led to a diagnostic review of the small bowel resection reported previously. It was agreed that it contained definite endometriosis. In addition, there were features of associated ESS. There were admixed endometrial glands within the sarcoma which were presumed to represent endometrioid differentiation within stromal sarcoma. Mitotic activity was infrequent (<5 mitoses per 10HPF). A focus of lymphovascular invasion was also noted in the review Figure .A final diagnosis of low-grade ESS with relatively prominent endometrioid glandular differentiation and associated endometriosis was given.This case is being highlighted to discuss its complexity and associated pitfalls. The case was discussed at a discrepancy meeting in the histopathology department and there was consensus that this case was challenging. None of the participants had come across such a case in their career. In addition to the rarity of the disease, incomplete clinical details on the request form (sent with specimen), without any suspicion of malignancy, from the surgeons contributed to the missed diagnosis. The presence of this information may have supported a diagnosis of malignancy at the time of the original pathology report. It was agreed that vigilance is required when dealing with extrauterine endometriosis and in particular when the stromal component shows exaggeration and prominent expansion.ESS is a rare tumour with about 80 cases reported so far ,11. A caThe development of malignancy in endometriosis was studied quite early. In 1925, Sampson  reportedAs seen from the case above, the diagnosis of low-grade ESS with prominent glandular differentiation is difficult, especially when the primary presentation is in the intestine. It can be confused with many other lesions, some of which will be discussed below.The main diagnostic difficulty in our case was the differentiation between endometriosis and ESS with extensive glandular differentiation. This is understandably difficult and some light is shed on this topic by a research article by McCluggage et al. . This arAnother important differential for our case was stromal endometriosis, which is known to simulate a sarcoma . StromalAggressive endometriosis is another entity commonly confused with extrauterine ESSs . This diOther differential diagnoses of primary extrauterine stromal sarcoma in the intestine include gastrointestinal stromal tumour (GIST) and Mullerian adenosarcoma with sarcomatous overgrowth. GIST is reported as the most prevalent stromal tumour in the intestine. It is important to differentiate ESS from GIST as the management route for both of these tumours is completely different. GISTs are well-circumscribed and have pushing borders in contrast to ESS which is invasive. Positive immunohistochemistry with c-kit is usually confirmatory for a diagnosis of GIST.A less common condition that can be included in the differential diagnosis is Mullerian adenosarcoma with sarcomatous overgrowth. The main histological clue to this diagnosis is the presence of different kinds of Mullerian epithelium. Other findings are periglandular cellular stromal condensation and extension of cellular stroma into the lumen of glands.The diagnosis of intestinal low-grade ESS with endometrioid glandular differentiation and associated endometriosis is difficult with a high likelihood of misdiagnosis and delayed diagnosis. A careful morphological and immunohistochemical examination is the key to the correct diagnosis.


A 6-year-old boy was referred because of a suspected recurrence of a growth hormone (GH)–producing pituitary macro-adenoma, which initially expanded from the sella turcica to the suprasellar and right parasellar region. Over the course of 2.5 years, the patient had undergone three transsphenoidal resections after which biochemical values normalized. At time of referral, the patient again experienced symptoms of localized headache and increasing GH and IGF-1 serum levels. Repeated MRIs were obtained under a dedicated pituitary imaging protocol, but paediatric neuroradiologists were unable to conclusively differentiate between recurring adenoma and post-operative granulation tissue.l-tyrosine ([18F]FET) PET has been described [18F]FET PET/MRI in our patient, in an attempt to confirm the suspected recurrence. [18F]FET is a non-natural amino acid transported by the amino acid transport system L (LAT), of which particularly the LAT1 isoform is expressed in a variety of tumour cells. A recent study in rat showed higher LAT1 gene levels in GH-producing pituitary tumour cells compared to normal pituitary tissue [As one report of the incidental finding of a macro-adenoma on 18F-fluoroethyl-y tissue .18F]FET uptake caudomedially of the right internal carotid artery . The uptake correlated with two nodular lesions of approximately 3 and 5 mm diameter , which were indeed progressive in size over time. This confirmed recurrent adenoma foci, and establishes the usability of [18F]FET PET/MRI for imaging of pituitary adenoma. Our patient was referred for radiotherapy, since a fourth resection was not considered possible.In our case, PET/MR-images revealed increased [


Paracoccidioidomycosis (PCM) is a systemic fungal infection, primarily affecting the respiratory tract. Extra pulmonary presentation is rare, representing less than 1% of cases (about 1 in every 200 cases).We present a case of a 73-year-old male with acute surgical abdomen secondary to Intestinal Paracoccidioidomycosis requiring intestinal resection and postoperative antifungal therapy.Intestinal Paracoccidioidomycosis represents a rare pathology with challenging diagnostic approach due to its frequency and nonspecific clinical manifestations. Extra pulmonary presentation is rare, but it should be considered in endemic regions. •Extrapulmonary presentation of PC it's a rare entity, reaching just 1% of the cases.•Fungal infection should be in the knowledge of general surgeon•IP, represents a surgical challenge, a multidisciplinary approach needs to be done in order to achieve good outcomes. Extra pulmonary presentation is rare, representing less than 1% of cases (about 1 in every 200 cases) 2After ethical and institutional approval, previous informed consent filled, following SCARE guidelines Physical examination was performed finding a painful abdomen with voluntary muscular defense with greater intensity on the lower quadrants. He had no fever or dyspnea. White-cell count was elevated with neutrophilia . Other checkups reported a non-inflammatory urinalysis, negative urine gram stain, arterial gases with compensated metabolic acidosis, without hyperlactatemia and a positive fecal occult blood test.Due to his medical history, a CT angiogram was performed disclaiming vascular pathology, no evidence of nephrolithiasis, urolithiasis, and appendicitis. Diverticular disease of the descending and sigmoid colon without signs of complication was reported, no signs of pneumoperitoneum, nevertheless inflammatory process of ascending colon associated to small bowel and ascending colon dilation with alteration of adjacent fat was documented . DespiteA right hemicolectomy with segmental jejunum resection, transverse ileum, and jejunum jejunal anastomosis were performed by an experienced general surgeon. Intraoperative findings showed a solid intraluminal lesion in the cecum of 7 cm (greater diameter), cecum walls of necrotic appearance, without evidence of perforation or free liquid in the peritoneal cavity. Additionally, a jejunal lesion comprising 80% of the small bowel's diameter was found at 110 cm from the Treitz ligament. and four lesions of 0,5 cm were found in the liver.Intensive Care Unit (ICU) was required during postoperative due to patients' need of vasopressor and mechanical ventilation. Once extubated and with no vasoactive support needed, the patient was transferred to an inpatient postoperative floor, in the next days developed dysphagia, abundant production through nasogastric tube, flats present but no feces, for which parenteral nutrition and postoperative ileus management was started. Despite adequate supportive care, there was no improvement, with increased drainage through nasogastric tube, pain, and distention. Revision of abdominal cavity was performed 8 days after first intervention finding 400 cc of hemoperitoneum and both anastomoses unscathed, due to impossibility of closure of abdominal wall patient required an additional procedure 4 days later to abdominal wall closure.Pathology reported ulcerated colonic and jejunal mucosa with presence of granulomas some of them with central necrosis, chronic inflammation that compromises subserosa immunohistochemistry reported Gomory + , Ziehl Neelsen - AE1/AE3 and CAM5.2 positive in cryptic epithelium confirming diagnosis of gastrointestinal Paracoccidioidomycosis.During hospitalization patient presented nosocomial pneumonia and needed percutaneous drainage of intra-abdominal collection both treated successfully with no further complications. After 56 days of inpatient care, he was discharged with antifungal extended therapy. His controls have been uneventful up to 6 months.3Paracoccidiodes brasiliensis. It was first described as a disseminated disease in Buenos Aires, Argentina in 1892 Paracoccidioides grows in the soil of humid regions with a medium to high rainy season, mild temperatures, and the presence of forests and rivers Paracoccidioidomycosis (PCM) is a systemic fungal infection generally self-limited to lung tissue caused by the dimorphic fungi Infection is acquired by arthroconidia  inhalation. Once the fungus invades the lungs in the yeast form, a non-specific inflammatory response is initiated and over 80% of cases are asymptomatic The extra pulmonary presentation is rare, representing less than 1% of cases (about 1 in every 200 cases) Gastrointestinal presentation is also infrequent, being the meninges, skin, bone, joints, mediastinum, and lymph nodes the most frequent involved organs There have been around 10 cases of abdominal Paracoccidioidomycosis reported in literature and given its extremely rare presentation, only clinical suspicion in endemic areas favors diagnosis Treatment includes monotherapy of amphotericin B, ketoconazole, fluconazole, or itraconazole PCM is a disease with high morbidity but relatively low mortality, relapses are common so it's important to receive and complete appropriate antifungal therapy 4Intestinal Paracoccidioidomycosis represents a rare pathology with challenging diagnostic approach due to its frequency and nonspecific clinical manifestations. Extra pulmonary presentation is rare, but it should be considered in endemic regions. Therefore, all cases should be published or added to scientific repositories to make data available regarding the management and outcomes for future reference and strengthening of knowledge.Not commissioned, externally peer-reviewed.Written informed consent was obtained from the patient for publi-cation of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ethical approval of institutional committee was made previous publication.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Felipe Giron, MD, MSc: Make substantial contributions to conception and design, acquisition of data, analysis and interpretation of data.Marco Vanegas, MD: Participate in drafting the article and revising critically for important intellectual content.Lina Marcela Rodriguez, MD: Participate in drafting the article and revising it critically for important intellectual content.Valentina Hernandez, MD student: Participate in drafting the article and revising it critically for important intellectual content.Carlos Eduardo Rey Chaves, MD: Make substantial contributions to conception and design, acquisition of data, analysis and interpretation of data.Jairo Ortega, MD: Give final approval of the version to be submitted and any revised version.Felipe Giron.None.Authors do not declare any conflict of interest.


We report a rare case of malignant phyllodes tumors (MPT) with partial response to apatinib.A 26-year-old woman had a palpable mass in her right breast for over a year. After resection, pathology indicated malignant phyllodes tumor. Eleven months after surgery, she underwent reoperation for a lung nodule, which demonstrated lung metastasis. She refused chemotherapy and was rehospitalized six months later due to leg pain. Pelvic mass biopsy revealed metastatic malignant phyllodes tumor. After concurrent chemoradiotherapy of the pelvic mass, multiple lung metastases emerged. Subsequent treatment with apatinib 500 mg/day resulted in a reduction in mass size and partial response. She survived for more than 8 months.The present case showed the potential therapeutic effects of apatinib in patients with MPT. Phyllodes tumors are rare fibroepithelial lesions and account for 0.3–1% of breast cancers . MalignaC-KIT or PDGFRA was detected by gene sequencing. Chemotherapy was recommended, but the patient refused it.On November 7, 2016, a 26-year-old Chinese woman was admitted to Drum Tower Hospital because of a palpable mass in the right breast that had been present for over a year. The patient underwent a right mastectomy and sentinel lymph node biopsy. Pathological and histopathological examination revealed a breast MPT measuring 17 cm in in maximum diameter. The lateral and basal margins were clear. Immunohistochemistry (IHC) showed Bcl2 ( +), P53 (+ + +), CD34 (Vessel +), Desmin (−), SMA (−), S100 (−), ER (−), PR ( ±), and HER2 (−). The Ki-67 proliferation index was 70%. On June 2, 2017, follow-up computed tomography (CT) showed a lesion in the left lung. The lesion was then resected by thoracoscopy. Pathological examination indicated metastatic MPT  50 Gy/10 fractions and planning gross tumor volume (PGTV) 70 Gy/10 fractions was prescribed concurrently with paclitaxel liposome 120 mg per week. The reset CT scan on January 31, 2018, showed that the real component of the mass decreased. The pain was significantly relieved from 6 to 1 on the pain scale, and the patient was able to walk freely after radiotherapy.Six months later, the patient returned with persistent pain in the left hip. Positron emission tomography-CT showed a pelvic mass with increasing 18F-FDG uptake. A percutaneous pelvic mass biopsy was performed, revealing a MPT metastasis. IHC showed PR (+), P16 (+ + +), ER (−), CD34 (−), CD117 (−), S100 (−), desmin (−), MA (−), and HER2 (−), and the Ki-67 index was 60%. Real-time RT-PCR analysis of the tumor specimens indicated that After approximately one month, the patient returned to the hospital with cough, chest tightness, wheezing, and slight hemoptysis. The CT scan revealed multiple lesions in both lungs Fig. , while tMPTs constitute 20% of all phyllodes tumors, are highly aggressive and have a tendency to form distal metastases . The aveCurrently, the treatment for MPT remains challenging, with no standard therapeutic regimen. According to the National Comprehensive Cancer Network (NCCN) guidelines, the recommendation for treatment of MPT is complete surgical resection with at least 1-cm margins without sentinel lymph node biopsy. The usage of radiotherapy has also recently increased. Nevertheless, the reduced local recurrence but lack of disease-free or OS advantage has been a limiting factor in conventional radiotherapy , 12. In Many pathways and molecules are related to the pathogenesis of phyllodes tumors. The expression of several biomarkers, including P53, C-KIT, CD10, and epidermal growth factor receptor, has been investigated . NeverthTumor angiogenesis plays a vital role in the initiation, progression and metastasis of tumors. Antiangiogenic therapies is a promising anticancer treatment strategy. Tyrosine kinase inhibitor (TKI) targeting VEGFR has led to remarkable advances in the treatment of many tumors. At least 20 VEGFR-TKIs  have been approved by the FDA, all TKIs have shown promising preclinical and clinical effectiveness against sarcoma. However, it is reported that the efficacy of apatinib is comparable or even superior to other TKIs . Its ant5. Nevertheless, it is unclear whether apatinib affects the treatment of phyllodes tumors. Sarcoma, a group of heterogeneous malignant tumors derived from mesenchymal tissue, is an indication of VEGF/VEGFR targeted therapy [Apatinib is a TKI that targets the VEGFR2 signal and exhibits a powerful antitumor effect in a variety of solid tumors , 19. It  therapy . Thus, b therapy . Two ret therapy , 23.C-KIT or PDGFRA, excluding the indication for imatinib, a C-KIT and PDGFRA inhibitor. Then, we performed real-time RT-PCR analysis, and the results of mRNA expression testing of BRCA1, TOP1, and RRM1 implied that the patient might be sensitive to paclitaxel, irinotecan, and gemcitabine. Then, we chose paclitaxel as a radiosensitizer for radiotherapy. The concurrent chemoradiotherapy effectively relieved the symptoms. However, the lesions in the lungs progressed. For systemic treatment, we referred to the treatment of sarcoma and used apatinib. Apatinib successfully reduced the mass size in the lungs. The survival time had exceeded 8 months since the treatment with apatinib, but she has since been lost to follow-up. There was no serious toxicity except for controllable and well-tolerated hemoptysis. Thus, the long-term efficacy of apatinib for phyllodes tumors can be expected.In this case, the patient had undergone surgeries at the primary site and for lung metastasis; however the disease continued to progress, and her physical condition deteriorated accordingly. The gene analysis showed no mutation in The present case showed the potential therapeutic effects of apatinib in patients with MPT. To our knowledge, this is the first report of apatinib in the treatment of phyllodes tumors. For advanced cases with multiple metastases, finding an effective targeted drug is very important for patient prognosis and quality of life. Further cohort and prospective trials are needed to identify a subset of patients suitable for apatinib in the clinical treatment of phyllodes tumors.


The benefit of immunochemotherapy in treating resectable locally advanced non-small cell lung cancer (NSCLC) is not well established. Here, we report a case of resectable stage III NSCLC treated with neoadjuvant immunotherapy combined with chemotherapy before surgery. A 61 years old man was admitted to our hospital due to paroxysmal cough and was diagnosed as squamous cell carcinoma T4N2M0 in the upper lobe of the right lung, which was locally advanced and resectable. He was treated with 3 courses of paclitaxel 250 mg intravenous (IV), carboplatin 0.65 g IV, and durvalumab 620 mg IV followed by thoracoscopic upper lobectomy and lymph node dissection. There was considerable regression of the tumor before surgery, and the patient achieved a complete pathological response after surgery. Our case study demonstrates the benefit of durvalumab and chemotherapy in the treatment of resectable locally advanced NSCLC. TheDespite curative resection in patients with NSCLC, 30–70% of patients will experience relapse resulting in death ,10. For 2A 61 years old man  score: 0–1) was admitted to our hospital on April 10, 2020, due to paroxysmal cough for 6 months. There was no complaint of having expectoration, hemoptysis, chest pain, hot flashes, and night sweats. The patient had a paroxysmal cough for 6 months, and he left it undiagnosed. On April 1, 2020, the patient underwent chest computed tomography (CT) examination as suggested by a local hospital physician. The CT scan reported a space of about 7.9 cm × 6.7 cm × 6.0 cm in the right upper lobe of the lungs. He was admitted to our hospital for treatment. He was a heavy smoker (20 cigarettes per day for 40 years) with no family history of cancer. The patient’s medical report during admission showed that the case was diagnosed as lung cancer .On physical examination during admission, there was no swelling of superficial lymph nodes in the whole province. During chest examination, the respiratory sounds of the right upper lung were diminished, and the respiratory sounds of the other parts of the lungs were clear. No dry and wet rales were found in both lungs. The thetumor marker levels in the serum were neuron-specific enolase (NSE) 12.5 ng/mL, squamous cell carcinoma antigen (SCC Ag) 1.7 ng/mL, and cytokeratin 19 fragments 15.8 ng/mL. On April 13, 2020, a chest CT examination showed bronchial obstruction in the right upper lobe surrounded with soft tissue mass, with a size of about 7.9 cm × 6.7 cm × 6.0 cm . Based oSleeve resection and lymph node dissection or neoadjuvant chemotherapy plus sleeve resection or radical radiotherapy was suggested to the patient. Based on the patient and his family’s consent, neoadjuvant immunotherapy combined with chemotherapy before surgery was considered. The first course of treatment was paclitaxel 250 mg intravenous (IV) + carboplatin 0.65 g IV and durvalumab 620 mg IV was given along with chemotherapy. On May 13, 2020, during re-examining the tumor markers, NSE was 13.8 ng/mL, SCC Ag was 1.4 ng/mL, and the cytokeratin 19 fragment was 1.9 ng/mL. The second course of treatment was carried out on May 14, 2020.On June 10, 2020, the patient was subjected to chest CT enhanced re-examination. The CT scan showed the presence of bronchial stenosis and occlusion in the right upper lobe along with soft tissue mass shadow with a size of about 5.0 cm × 3.2 cm × 2.8 cm . The uppThe clinical efficacy evaluation performed on July 9, 2020, with fiberoptic bronchoscopy showed that after treatment with neoadjuvant immunotherapy and chemotherapy, the right upper lobe of the right lung, the bronchus of the right upper lobe, was unobstructed, and tracheobronchitis occurred . In addiAfter considerable tumor regression, thoracoscopic upper lobectomy and lymph node dissections were performed on July 14, 2020. During the surgery, the tumor subsided >1 cm distal to the bronchial opening of the right upper lobe. No cancer was observed during the surgery. On postoperative pathological reports, the size of the lung tissue was 12 cm × 10 cm × 5.5 cm, and the size of the tumor bed was 3 cm × 2 cm × 2 cm. Under a microscope, necrosis, histiocyte reaction, cholesterol crystal, and inflammatory cell reaction were observed. No residual tumor cells were found, in line with the post-treatment reaction . ImmunohInformed consent: Informed consent has been obtained from all individuals included in this study.Ethical approval: The research related to human use has been complied with all the relevant national regulations, institutional policies and in accordance with the tenets of the Helsinki Declaration, and has been approved by the author's institutional review board or equivalent committee.3This case report describes a case with resectable NSCLC treated with neoadjuvant chemotherapy (paclitaxel + carboplatin) + immunotherapy  followed by surgery.N = 22), the major pathological response (MPR) in the resected tumor was 45%, and recurrence-free survival was 73% after 18 months of surgery [Neoadjuvant chemotherapy combined with durvalumab achieved a satisfactory CPR demonstrating preliminary promise for this treatment strategy. These findings are consistent with the preliminary results of previous studies ,18,19,20N = 46) before surgery [N = 30) [Further, a few more studies were carried out to explore the effect of immunotherapy combined with chemotherapy at neoadjuvant settings ,19,20. A surgery ,19. Arou surgery . Yet anoIn phase 3 PACIFIC trial, patients with stage III unresectable NSCLC treated with durvalumab in adjuvant settings showed significantly longer progression-free survival and overall survival than patients treated with placebo ,15. PACIAfter considerable regression of the tumor in the patient, we opted for thoracoscopic upper lobectomy and lymph node dissection as the pulmonary edema was mild and the separation of tissue space was not difficult. In the neoadjuvant chemotherapy era, sleeve resection or lobectomy plus bronchoplasty was a reasonable surgical plan in the case of tumor growth and infiltration to the bronchial orifice before neoadjuvant therapy. At present, there is no definite conclusion about the clinical scope of lobectomy. However, in this case, we performed routine lobectomy and final pathological examination of the frozen bronchial resection edge. As the resected bronchial margin was negative for the tumor, we did not perform sleeve resection of the upper lung.Further, there is no clear guidance in clinical practice for postoperative adjuvant treatment in patients with resectable stage III NSCLC. However, based on the study NADIM trial, we can consider immunoadjuvant therapy for 1 year after surgery or conventional adjuvant chemotherapy for 4 courses .Further, based on the results of preliminary studies and our case study, it is still unclear whether neoadjuvant immunotherapy combined with chemotherapy can improve long-term patient prognosis. Hence, long follow-up results of these studies are warranted to confirm the efficacy and safety of neoadjuvant immunotherapy combined with chemotherapy in patients with resectable stage III NSCLC.4In this study, patients with resectable stage III NSCLC treated with neoadjuvant chemoimmunotherapy achieved a complete pathological response, thus providing clinical evidence for future research. However, advanced phase 3 studies with long-term follow-up are imperative to provide further evidence for the chances of cure for a potentially lethal locally advanced resectable NSCLC.


Teaching point: Extrahepatic portosystemic shunt is a rare congenital condition, associated with other congenital anomalies, for which prompt radiological diagnosis and treatment are important to prevent complications. A four-month-old female patient with prenatal diagnosis of Tetralogy of Fallot underwent abdominal ultrasound for rising bilirubin levels after cardiosurgery.Figure A, arrow). No portal vein Doppler signal could be detected in the liver. Pulsed-wave Doppler in the hepatic and splenic veins showed the same flow pattern , indicating a communication between the systemic and splanchnic veins. A contrast-enhanced computed tomography (CT) of the abdomen was performed to demonstrate properly the anatomy of the splanchnic veins. It confirmed a venous malformation, with confluence of the splenic and superior mesenteric vein into a common trunk that drained directly into the right atrium, also named “Abernethy malformation type 1b” . The patient eventually died a month after surgery without being treated for the malformation.It revealed homogeneous liver parenchyma, normal gallbladder, and no dilatation of the biliary ducts. However, the portal vein could not be demonstrated. Where we would normally expect the intra-hepatic portal vein there was a hyperechoic smooth band of 3–4 mm  is a rare malformation in which the portomesenteric blood drains directly into the systemic circulation. CEPS is classified into two types. In type 1 malformations there is a congenital absence of the portal vein. These are further divided into those in which the splenic and superior mesenteric vein drain separately into a systemic vein (type 1a) and those in which the two veins form a common trunk that drains into the systemic circulation (type 1b). In contrast, the portal vein is preserved in type 2 malformation, but some of the portal flow is bypassed to the systemic circulation through a side-to-side shunt [Clinical manifestations are related to abnormal hepatic development, the portosystemic shunt and associated congenital anomalies, for instance congenital heart defects. The presence and extent of complications, such as portosystemic encephalopathy, and associated anomalies determines the prognosis .Usually, CEPS is discovered incidentally on routine ultrasound (US) or in a symptomatic child with nonspecific liver disease. Absence of the portal vein is most often demonstrated. In contrast to other causes of spontaneous shunts, patients with CEPS do not have imaging features of portal hypertension . CT angiIt is essential to determine the type of shunt because treatment options are totally different . Eventua