Blastomycosis is a systemic mycotic infection caused by dimorphic fungi. The disease is rare in cats, and reports on imaging findings with central nervous system (CNS) involvement are limited. Magnetic resonance imaging (MRI) was performed antemortem in three feline patients. Imaging findings that may allow prioritization of intracranial blastomycosis over other differential diagnoses included focal or multifocal intra-axial mass lesions with dural contact, lesion hypointensity on T2-weighted images and diffusion-weighted imaging/apparent diffusion coefficient map (DWI/ADC), strong and homogeneous contrast enhancement of the lesion(s), concurrent meningeal enhancement, marked perilesional edema and mass-effect, and ocular abnormalities. One cat was managed successfully and had a recurrence of CNS blastomycosis more than 4.5 years after the initial diagnosis. Repeat MRI at that point revealed both new and persistent (chronic) abnormalities. Blastomyces dermatitidis and Blastomyces gilchristii have an endemic distribution in the Mississippi, Missouri, and Ohio River valleys, the mid-Atlantic states, and some Canadian provinces with sporadic cases reported in other regions  included asymmetric hydrocephalus with marked contrast enhancement of the ventricular lining (Computed tomography (CT) and magnetic resonance imaging (MRI) findings in dogs with intracranial blastomycosis are variable and include solitary or multifocal mass lesions that can be intra- and/or extra-axial and may extend from the nasal cavity or orbit into the cranial vault, perilesional edema, mass–effect and brain herniation, meningeal thickening and contrast enhancement, and ventricular lesions characterized by ventriculomegaly, ependymal or periventricular contrast enhancement, and evidence of increased intracranial pressure –19. Repor lining .In this case series, we report the MRI findings in three domestic cats diagnosed with CNS blastomycosis. Case 2 was included in a previous publication on ocular blastomycosis , but dia®, Bayer Schering Pharma; Case 3: 0.1 mmol/kg IV gadodiamide, Omniscan®, GE Healthcare). The MRI protocols varied slightly between the cases. The sequences acquired in all patients included sagittal T2-weighted spin echo (SE); transverse T2-weighted SE, T1-weighted SE, proton-density (PD)-weighted SE, T2-FLAIR, and T2*-weighted gradient recalled echo (GRE); transverse postcontrast volume interpolated GRE images with fat saturation (FatSat); and postcontrast transverse, dorsal FatSat and sagittal T1-weighted SE images. Diffusion-weighted imaging (DWI) and apparent diffusion coefficient (ADC) maps were available for review for the second scan in Case 1 and Case 3.The MRI scans were performed using a 1.0 T MRI system  (Case 1 first scan and Case 2) or a 1.5T MRI system  (Case 1 second scan and Case 3). Intravenous contrast medium was administered in all cases  was performed and was highly positive (above the limit of quantification). The clinical signs and lung nodules had improved with treatment . The reported tonic–clonic seizures occurred after the most recent antifungal treatment had been administered. At presentation, no abnormalities were noted on physical and neurologic examination.An 11-year-old male castrated 5.7-kg domestic shorthair cat presented to the internal medicine service after experiencing two seizures (believed to be tonic–clonic). The medical record information prior to this presentation is limited. The patient was feline immunodeficiency virus (FIV) positive and had been seen at the referring veterinarian 3 months prior with a presenting complaint of fever and ataxia. Pulmonary nodules were identified on thoracic radiographs at that time. A diagnosis of systemic blastomycosis was made based on fine-needle aspiration (FNA) of an enlarged popliteal lymph node. A *-weighted images, and was strongly homogeneously contrast enhancing. There was moderate indistinct perilesional T2 hyperintensity, consistent with perilesional edema. A second well-circumscribed ≈0.4-cm diameter T2 hypointense and T1 isointense lesion was associated with the right cerebellar hemisphere and exhibited moderate ring enhancement following contrast medium administration. A third ≈0.2-cm diameter ring-enhancing lesion was noted in the cerebellar vermis, not seen on precontrast images. There was no evidence of perilesional edema associated with the cerebellar lesions. There was no evidence of meningeal enhancement or any abnormalities of the ventricular system, skull, nasal cavity, paranasal sinuses, tympanic bullae, orbits, ocular structures, and soft tissues of the head and cranial neck including regional lymph nodes.On MRI of the brain, three separate intra-axial lesions were identified . A roundBlastomyces urine antigen EIA was moderately positive (3.04 ng/ml).A cisternal cerebrospinal fluid (CSF) tap was obtained and yielded a diagnosis of probably iatrogenic hemorrhage without other cytologic abnormalities. A Corticosteroid treatment was initiated  for treatment of perilesional edema and to help manage the inflammatory effects caused by fungal die-off. The patient was also started on phenobarbital (2.5-mg/kg PO BID of 20-mg/ml suspension), levetiracetam (20-mg/kg PO of 100-mg/ml suspension q 8 h), and fluconazole (50-mg/head PO q 12 h). Amphotericin B was discontinued. Phenobarbital was discontinued the following day due to the patient being very lethargic.Blastomyces urine antigen EIA was greatly improved but still mildly positive (0.73 ng/ml).The cat showed improvement over the next months and was considered clinically normal aside from an occasional cough and unrelated chronic gingivitis 2 months after the MRI examination . The previously seen pulmonary abnormalities had resolved. There was no recurrence of seizures, and no other neurologic abnormalities were reported. A Blastomyces urine antigen EIA was still mildly positive after 7 and 9 months. Fluconazole was discontinued 15 months after the initial diagnosis (12 months after the first MRI examination) based on 2 consecutive negative Blastomyces urine antigen EIA tests.The patient remained neurologically normal. Levetiracetam was gradually tapered from 5 to 8 months after the initial diagnosis without recurrence of seizure activity. A The cat represented 3 years later  to the emergency service for a 24-h history of circling to the right and behavior changes. On neurologic examination, the patient was circling to the right and had a bilaterally absent menace response. Possible hyperesthesia of the head was also noted. Neurolocalization was to the right forebrain. Thoracic radiographs did not reveal any significant abnormalities.*-weighted images and had mild rim contrast enhancement. The lesion within the cerebellar vermis was no longer visible. An incidental small amount of fluid was noted within the bilateral tympanic bulla. The remaining structures of the head and cranial neck remained normal.Magnetic resonance imaging was repeated. There was a new finding of an ≈1.2 × 1.4 cm ovoid mass in the right temporal lobe  which waBlastomyces urine EIA test was positive (4.24 ng/ml), and fluconazole treatment was continued. The patient improved, and the client reported the cat as being back to normal 5 weeks after the second MRI examination. On neurologic examination at that time, menace response was noted in both eyes. The pupillary light responses were decreased. Additional findings during this visit included weight loss, hypoalbuminemia, anemia, a soft heart murmur, and an irregular heartbeat. The patient represented for a work-up of these new problems to the internal medicine service 2 weeks later and at that point was diagnosed with large cell gastrointestinal (GI) lymphoma. Chemotherapy was started and initially well-tolerated. The owners did not report a recurrence of neurologic abnormalities, and the physical examinations performed by the oncology service over the next weeks did not reveal any obvious neurologic abnormalities. A full neurologic exam was not performed. Urine blastomycosis antigen tests were also not repeated during that time due to the inability to acquire sufficient urine samples. The patient started declining ≈2 months after the diagnosis of GI lymphoma and was euthanized 3 months later. An autopsy was not performed. The overall survival time after the initial diagnosis of systemic blastomycosis was ≈5 years.The patient received a tapering dosing regimen of prednisolone . Fluconazole treatment was resumed empirically (50-mg/head PO q 12 h) while awaiting urine antigen testing results. A repeat A 3-year-old 4.7-kg female spayed domestic shorthair cat presented to the emergency service after a mass was noted associated with the conjunctiva of her right eye. Two weeks prior she had been seen by the ophthalmology service for uveitis and secondary glaucoma of the right eye . Physical examination findings and a complete blood count (CBC) performed at that point were normal. The infectious disease testing for feline leukemia virus (FeLV), FIV, and toxoplasmosis were negative. The patient was managed with topical medications. At the current presentation, the cat had worsened uveitis OD and blindness in both eyes  with a fixed dilated pupil of the left eye . Thoracic radiographs revealed a single ≈2.5-cm diameter ill-defined soft tissue opacity mass associated with the left caudal lung lobe. A fine-needle aspiration of the conjunctival mass was performed and yielded a diagnosis of blastomycosis. Due to concern for intracranial involvement, MRI of the brain was performed .There were marked abnormalities of the right eye including severe thickening and contrast enhancement of the periocular tissues, thickening and enhancement of the wall of the globe, retinal detachment, diffuse heterogeneous alteration in signal intensity of the ocular structures with abnormal contrast enhancement, and multifocal susceptibility artifacts, mostly in the posterior chamber, consistent with hemorrhage. There was diffuse thickening of the right optic nerve (up to 5 mm) and marked enlargement of the optic chiasm (1.3 cm). Two additional intracranial lesions were noted. A round 0.5 cm diameter intra-axial nodule was associated with the caudal aspect of the right frontal lobe. An irregularly shaped ≈0.8 × 0.7 cm nodule was associated with the left parietal lobe. This lesion was in contact with the falx and meningeal surface of the brain but formed an acute angle with both. Both lesions were iso- to mildly hypointense to gray matter on both T1- and T2-weighted images. Bordering the lesions and extending extensively throughout the cerebral white matter bilaterally, there were parenchymal hyperintensities consistent with vasogenic edema. There was evidence of increased intracranial pressure characterized by attenuation of the CSF signal in the cerebral sulci, compression of the lateral ventricles, a mild midline shift, and caudal transtentorial and foramen magnum herniations. Following contrast medium administration, the right globe, abnormal tissues within the right orbit, right optic nerve, optic chiasm, and intracranial nodules showed strong and homogeneous contrast enhancement. There was no evidence of abnormal meningeal contrast enhancement and no evidence of a dural tail sign adjacent to the brain lesions. Moderate enlargement of the right and mild enlargement of the left medial retropharyngeal lymph nodes were noted.The patient was euthanized, and an autopsy was performed, confirming the clinical diagnosis of blastomycosis. There was marked pyogranulomatous panophthalmitis of the right eye with involvement of the optic nerve and the optic chiasm. Both cerebral nodules were intra-axial in location, and a diagnosis of a marked pyogranulomatous meningoencephalitis with intralesional yeasts was made. Additional lesions were found in the lungs (as suspected based on radiographs), the thoracic lymph nodes, and the fourth and fifth digits of the right front limb.An estimated 7-year-old 6.1-kg male castrated domestic shorthair cat presented to a local emergency clinic for head pressing. Complete blood count and blood chemistry panel were normal, and FeLV/FIV testing was negative. The patient was referred for progressive neurologic abnormalities. Upon presentation, the cat was bright, alert, and responsive. One hind limb had been amputated previously for unknown reasons. Neurological deficits included a lack of menace response of the left eye (OS) and intermittent loss of the right eye (OD), bilateral (OU) loss of direct and indirect pupillary light reflexes, and circling to the right. Neurolocalization was to the right forebrain and bilateral optic nerves/optic chiasm. A mild diffuse bronchointerstitial pattern was identified on thoracic radiographs.*-weighted images, and was hypointense on both DWI and ADC maps. There was marked perilesional T2 hyperintensity extending along the white matter tracts of the right cerebral hemisphere, consistent with vasogenic edema, resulting in compression of the right lateral ventricle, a midline shift toward the left, caudal transtentorial, and mild foramen magnum herniations. Following contrast medium administration, the mass showed strong and fairly homogeneous contrast enhancement. The majority of the lesion was centered on brain parenchyma, with acute angles between the ventral, rostral, and caudal aspects of the mass and the calvarium. At the dorsal aspect of the mass and best seen on thin section postcontrast T1-weighted FatSat GRE images, there was mild focal thickening and contrast enhancement of the pachymeninges. A small volume of fluid was present in the nasopharynx, considered incidental. There were no abnormalities of the skull, nasal cavity, paranasal sinuses, tympanic bullae, orbits, ocular structures, and soft tissues of the head and cranial neck including lymph nodes.On MRI examination of the brain, there was an ≈1-cm diameter mass associated with the periphery of the right temporal lobe, in contact with the dural surface . The lesA CSF tap was not performed. The patient was administered mannitol (0.5 g/kg IV) and dexamethasone sodium phosphate (0.1 mg/kg IV). Blood and urine samples were submitted for cryptococcosis and blastomycosis testing, respectively. The patient was discharged the following day on prednisolone (2-mg/head PO q 24 h) and levetiracetam (20-mg/kg PO of 100 mg/ml suspension q 8 h).Blastomyces urine antigen EIA was positive (6.11 ng/ml). The patient was prescribed fluconazole (50-mg/head PO q 24 h). Two days after the start of treatment, the owner reported sudden deterioration of the patient followed by death. An autopsy was not performed.A cryptococcus antigen latex test was negative. A Inflammatory and neoplastic etiologies account for the majority of brain diseases in cats , 22, andEven though the imaging findings in the three cats included in this case series differed, some commonalities were observed. All animals had focal or multifocal mass lesions associated with the forebrain. Contact with the brain surface was common, resulting in difficulty classifying a lesion as intra-axial vs. extra-axial in some instances. Ultimately, intra-axial lesion location was considered most likely in all cats based on lesion centering on brain parenchyma, infiltrative rather than expansile growth of the mass, the presence of additional intra-axial lesions in the majority of cases including cerebellar lesions in one cat, and the acute rather than the obtuse angle formed between the periphery mass and the skull . An autopsy was only performed on one cat, confirming the intra-axial location of the lesion. Difficulty in distinguishing intra- from extra-axial lesion location was mentioned in a previous study describing CT findings in a cat with an intracranial blastomycosis granuloma  and has *-weighted GRE images which were not seen in the cases described here. CNS lymphoma has a variable imaging appearance in cats, with intra- and/or extra-axial masses of varying intensity and contrast enhancement patterns. While T2 hyperintensity is a more common feature and T2 hypointensity has not been reported, T2 isointensity is possible   was not performed. This is representative of clinical veterinary practice, where organism identification through cytologic or histopathologic evaluation is preferred over serology due to the associated cost for the client . The idele cause , 36. Bec species . In peoprichium) , 38. Howrichium) , 40. IntIn summary, the imaging features that may suggest blastomycosis over other intracranial diseases in cats include focal or multifocal intra-axial mass lesions with dural contact, hypointensity on T2-weighted images and DWI/ADC, strong and homogeneous contrast enhancement of the lesion(s), concurrent meningeal enhancement, marked perilesional edema and mass-effect, and ocular abnormalities. Future studies with larger case numbers are warranted to confirm these findings and identify possible other imaging manifestations of CNS blastomycosis in feline patients.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.Ethical review and approval was not required for the animal study because it is not required for retrospective case reports. Written informed consent was obtained from owners for participation of their animals.SH identified the cases to be included in the study, performed the image evaluation, and drafted the initial manuscript. JM and HS assisted with the medical record review of the patients and reviewed and revised the manuscript. All authors contributed to the manuscript's final revision and approved the 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.


Spontaneous isolated superior mesenteric artery dissection is a very rare vascular disease that involves the superior mesenteric artery or its branches, with an incidence as low as 0.08%. The majority of cases occur in patients of Asian descent. Due to advances in imaging modalities, particularly abdominal computed tomography angiography, the diagnosis of this disease has been increasing. Herein, we present a rare case of spontaneous isolated superior mesenteric artery dissection with thrombosis in a young male patient with no past medical history. The importance of this disease as a differential diagnosis for acute abdominal pain is emphasized. Conservative management, including bowel rest, intravenous fluid therapy resuscitation, and pain management, was started. Due to the persistence of the symptoms, CTA of the abdomen was performed, which confirmed the dissection in the proximal part of the SMA (A 43-year-old Caucasian man, without known medical history, presented with sudden-onset abdominal pain in the epigastric area radiating to the back, associated with nausea and nonbilious vomitus. The remaining review of systems was negative. The patient denied history of abdominal surgeries, and the family history was negative for coagulopathy and connective tissue disorders. He also reported 20 pack-years of tobacco smoking but denied alcohol and substance abuse. Vitals were unremarkable on admission and throughout the course of the hospital stay. An abdominal exam was negative for rigidity, tenderness, and bruits. Laboratory measures, including troponin, serum lipase, lactic acid, lactate dehydrogenase, and a lipid panel, were monitored and remained unremarkable. Computed tomography of the abdomen with intravenous contrast revealed a low-density related proximal aspect of SMA, 2.5 cm from the origin of the artery, suspected for SMA dissection the SMA A, with a the SMA B.Figure The patient was placed on intravenous heparin, and a vascular surgery consult was obtained. The intact flow to distal segments of the SMAs led to the recommendation of ongoing conservative management with anticoagulation. Serial lactic acid and lactate dehydrogenase levels were obtained to monitor for complications, including bowel ischemia, and remained unremarkable. After 48 hours of treatment, the patient’s symptoms resolved, and he was able to tolerate oral intake. The patient was discharged on apixaban, at 10 mg twice daily for 1 week, followed by 5 mg twice daily, for the duration of 3 months. A 3-month follow-up CTA of the abdomen revealed proper remodeling of the SMA and resolution of the dissection . The patVascular dissection occurs when a small tear in the intimal lining of a vessel exposes the deeper layers of an artery, and pressure forcibly separates these layers.Although conservative management is successful in many patients with SISMAD, operative or endovascular therapy is sometimes necessary despite the demonstration of flow in the distal vessels. Symptomatic patients treated conservatively are at risk of late secondary intervention, which occurs relatively often; therefore, long-term follow-up with imaging is warranted.•SISMAD is a rare etiology of acute abdominal pain, with no established management guideline.•Although a conservative approach with anticoagulants is successful in many patients with SISMAD and thrombosis, it occasionally requires operative or endovascular therapy.•Follow-up imaging with abdominal CTA might be useful to assure artery remodeling.According to the rarity of the condition, SISMAD often is not considered in the differential diagnosis for acute abdominal pain, out of proportion to the physical examination. CTA is a valuable imaging modality in these patients. Further studies are warranted to establish guidelines for the management and follow-up of SISMAD patients with thrombosis.


Idiopathic intracranial hypertension (IIH) is defined as raised intracranial pressure of unknown etiology. Looking for underlying causes needs to be undertaken before the diagnosis is confirmed and managed accordingly. We are reporting a rare and unique case of a patient with the clinical diagnosis of idiopathic intracranial hypertension or pseudotumor cerebri. She is not obese and not taking any predisposing medication. Her first magnetic resonance imaging (MRI) and magnetic resonance venogram (MRV) were reported as showing left transverse sinus thrombosis. She was treated with anticoagulation. Her final diagnosis of left transverse sinus compression by a meningioma en plaque (MEP) was finally made with a repeat MRI. A high index of suspicion of unusual causes is important when IIH presents in an atypical patient or with an atypical presentation. The prognosis and management of real IIH and raised intracranial pressure due to other causes are different. Idiopathic intracranial hypertension (IIH), which in the past was called pseudotumor cerebri, is defined as increased intracranial pressure (ICP) of unknown cause. All other causes of raised ICP need to be excluded before the diagnosis of IIH can be confirmed . It is cThe patient is a 42-year-old female with a known history of long-standing migraine without aura. She first presented to her primary care physician with a four-month history of a new kind of headache that was different from her previous migraines. Her new headache is mainly positional and occurred and worsened when lying down, coughing, sneezing, and exertion. She also complained of pulsatile tinnitus and intermittent blurring of vision with straining. She was not overweight, with a body mass index of 22. She was not taking any medication on a regular basis or recently, including tetracycline or vitamin A supplements. Because of her visual symptoms, she was evaluated by an ophthalmologist who found the presence of mild bilateral papilledema with normal visual acuity. The clinical diagnosis was idiopathic intracranial hypertension (IIH) or pseudotumor cerebri. Magnetic resonance imaging (MRI) of the head revealed left transverse sinus thrombosis Figure .Magnetic resonance venogram showed left transverse sinus occlusion Figure .She was started on treatment with dabigatran orally and referred to see a neurologist.When she was seen in neurology, her symptoms and her exam findings were similar. Her medication list included dabigatran, varenicline, Flexeril, and verapamil. We agreed with the diagnosis of IIH-like clinical presentation secondary to left transverse sinus thrombosis. We recommended continuing with the dabigatran therapy and repeating the MRI head in three months.The follow-up MRI three months later unexpectedly showed an en plaque meningioma (MEP) with hyperostotic growths compressing a long segment of the transverse sinus Figure .The final diagnosis was occlusion of the left transverse sinus due to an underlying meningioma en plague and its accompanying hyperostosis. Her intracranial hypertension was clearly not idiopathic but secondary to left transverse sinus occlusion secondary to external compression.Idiopathic intracranial hypertension (IIH), which, in the past, was called pseudotumor cerebri, is defined as increased intracranial pressure (ICP) of unknown cause. All other causes of raised ICP need to be excluded before the diagnosis of IIH can be confirmed . The undBased on her clinical features, with the exception of her normal BMI and lack of iatrogenic etiologies, her presentation was consistent with IIH. Our patient’s initial presentation is quite classic for IIH, with the exception that she is not obese and not on any medication associated with IIH. Her MRI head with MR venogram did show findings compatible with left transverse sinus thrombosis. Cerebral venous sinus thrombosis has different clinical presentations, and one type of presentation is like IIH. That is the reason why venographic studies are indicated in patients with a clinical diagnosis of IIH . Our patMeningiomas are benign central nervous system tumors originating from the meninges' arachnoid cap cells. Clinical manifestations of the disease are determined by the location as well as the size of the mass. While most meningiomas are asymptomatic, symptomatic cases exhibit upper motor neuron signs as well as symptoms concomitant with increased intracranial pressure  . MEP refOur patient’s situation is unique in its rarity as well as its manifestation. Knowledge of the atypical manifestation may lead to a broader and more complete differential diagnosis when assessing subtle MEP or for clinical presentations of IIH. The knowledge may allow for a faster diagnosis and prompt management.In order to make a definitive diagnosis of idiopathic intracranial hypertension (IIH) or pseudotumor cerebri, other causes need to be considered and ruled out. The commonest known cause is dural sinus thrombosis, which requires specific treatment with anticoagulation on its own merit. Our case demonstrates a rare cause, i.e., transverse sinus occlusion due to external compression by an MEP, which is difficult to be diagnosed with neuroimaging studies. A high index of suspicion for unusual causes of sinus occlusion is necessary for earlier diagnosis and appropriate management and therapy. This often requires a good clinical neurologist and a competent neuroradiologist.


Synovial sarcoma usually presents in the lower extremities and metastasizes to the lungs; however, unusual patterns of recurrence can occur. For patients with recurrent synovial sarcoma to a proximal peripancreatic lymph node, a pancreaticoduodenectomy or Whipple procedure is the best option for a cure. Lymph node metastasis from synovial sarcoma is exceptionally rare, and data guiding the use of the Whipple procedure for curative resection of metastatic synovial sarcoma are even more sparse. In this report, we describe the management of a patient with metastatic synovial sarcoma to a proximal peripancreatic lymph node with a pancreaticoduodenectomy. Synovial sarcoma is an aggressive mesenchymal neoplasm that metastasizes in 50% of patients . AlthougAn otherwise healthy 42-year-old woman was first found to have a 3.7-cm synovial sarcoma on workup of a lump she found with easy local bruising on the medial aspect of her left upper thigh. A core needle biopsy demonstrated a markedly cellular neoplasm consisting of round and spindle-shaped cells expressing BCL2, patchy CD99, and focal cytokeratin by immunohistochemistry; evidence of SS18 gene rearrangement by break-apart fluorescence in situ hybridization confirmed the diagnosis of monophasic synovial sarcoma. Once radiographic imaging showed no evidence for distant metastasis, the patient underwent a wide local excision yielding a margin-negative 4.5-cm tumor with 25 mitoses per 10 high-power microscopic fields, consistent with FNCLCC grade 3, pT1 disease. She was recommended to undergo adjuvant radiation to reduce the risk for local recurrence, but no systemic therapy was indicated since her tumor was less than 5 cm. She received 31 fractions of 57 Gy for adjuvant radiation. She unfortunately developed severe dysesthesia and motor weakness in the left leg following her operation that improved with physical therapy.After a year and a half, surveillance cross-sectional imaging revealed a new 4.3-cm mass involving the head of the pancreas without any pancreatic ductal dilation or parenchymal atrophy Figure .The patient had also developed new back pain and was referred for endoscopic biopsy, which revealed recurrent synovial sarcoma. She completed four cycles of neoadjuvant adriamycin and ifosfamide ending short of the intended six cycles due to intolerable nausea with vomiting as well as anemia requiring transfusion. On her last preoperative cross-sectional scan, her pancreatic mass had responded down to a size of 2.5 cm. The patient was taken for pancreaticoduodenectomy. Intraoperatively, a large volume of chylous fluid was encountered during the dissection that was controlled with clips and suture ligatures. The patient progressed well postoperatively and was discharged home with her surgical drain on postoperative day 6. Histologic examination of the pancreaticoduodenectomy specimen was notable for a neoplastic proliferation of predominantly spindle-shaped cells, involving pancreatic parenchyma and peripancreatic soft tissue (Figure The diagnosis of metastatic synovial sarcoma was confirmed by immunohistochemistry using an SS18-SSX fusion-specific antibody . There wThis case represents an unusual circumstance where a patient with a relatively confined, small synovial sarcoma developed a metastasis to a peripancreatic lymph node. Of the soft tissue sarcomas, synovial sarcoma is an intermediate- to high-grade malignancy for which five-year survival is noted in 36-76% . Althoug At the time of resection, there was extensive lymphatic drainage noted in the process of mobilizing the Whipple specimen. Some chylous fluid can be encountered as part of the Whipple pancreaticoduodenectomy, but a substantially greater volume of lymph was appreciated during the dissection for this patient, which is typically indicative of lymphatic obstruction. This observation in tandem with the odd pattern of metastasis in this patient may bear some relevance; however, this has been poorly defined in prior reports.Pancreatectomy for metastatic disease is a rare but previously described indication warranted when the pancreatic duct is suspected to be involved by disease. Only seven cases of metastatic synovial sarcoma to the pancreas have been previously reported Table . For perPancreatic metastasis from primary synovial sarcoma is exceedingly rare but manageable by pancreatectomy. If the tumor biology is not indicative of aggressive pathology, the rare event of lymph node metastasis may occur in patients with atypical lymphatic anatomy and may also be associated with higher volume chylous drainage noted during the surgical dissection. Further work is needed to better characterize the pathophysiology for uncommon patterns of metastasis as well as the indications of pancreatic metastasectomy.


Retinal vascular events may occur as rare complications of glaucoma procedures due to various factors, including exacerbation of ischemia in patients with pre-existing vascular comorbidities, toxic effect of mitomycin-C, and decompression retinopathy. We present the case of a 47-year-old hypertensive male who underwent trabeculectomy for advanced glaucoma in his right eye. At 3 weeks postoperatively, he presented with a drop in visual acuity to light perception with a spike in intraocular pressure. On examination, there was increased bleb vascularity as well as rubeosis. Fundoscopy revealed findings consistent with both central retinal artery occlusion and central retinal vein occlusion. Combined central retinal artery and vein occlusion is a rare retinal vascular condition. Neovascular glaucoma can occur as a sequelae of the ischemic process in the retina. Despite treatment, there is a poor visual prognosis, with the affected eye usually becoming blind from optic atrophy and neovascularization. Retinal vascular occlusions after intraocular procedures are rare. Various forms of retinal artery and vein occlusions have been reported following trabeculectomy. The exact mechanism behind their occurrence is unknown, but it is theorized that intraoperative pressure changes occurring intraoperatively and postoperatively act as a trigger for vascular events, particularly in patients with predisposing cardiovascular conditions .Combined central artery and vein occlusion (CCRAVO) is a particularly rare retinal vascular condition combining the clinical features of central retinal artery occlusion (CRAO) and central retinal vein occlusion (CRVO). Only a small number of cases have been reported, thus the clinical course and management have not been well established. CCRAVO has been reported in association with many conditions, including systemic lupus erythematosus, orbital inflammatory disease, Behcet’s syndrome, posterior scleritis, optic neuritis, leukemia, lymphoma, homocysteinemia, retrobulbar injection, and trauma. The condition is usually associated with a poor visual prognosis .To the best of our knowledge, this is the first reported case of CCRAVO occurring as a complication of glaucoma filtering surgery, highlighting the diagnosis and management of this rare complication. A 47-year-old male with a prior history of hypertension was being managed as a known case of juvenile open angle glaucoma on both eyes. The best corrected visual acuity (BCVA) on the right eye was hand motions, with an intraocular pressure (IOP) of 26 mmHg and optic disc examination and perimetry results consistent with advanced glaucoma. The left eye had a vision of no light perception from absolute glaucoma. Preoperative eye examination was significant for glaucomatous optic neuropathy with advanced neuroretinal rim thinning and disc pallor, as demonstrated by optical coherence tomography (OCT) of the optic nerve head Figure 1 . The antAt 3 weeks postoperatively, the patient presented with decreased vision in the right eye. The BCVA dropped to light perception. On biomicroscopy, fine rubeosis on the pupil edge, iris surface, and angles were identified  showed increased reflectivity and thickness of the inner retinal layers with posterior shadowing on the outer retinal layers. There was also minimal cystoid macular edema at the fovea Figure 4 .Fundus fluorescein angiography (FFA) done at presentation Figure 5  showed dThe clinical features as well as results of the FFA and OCT confirmed the diagnosis of CCRAVO with secondary neovascular glaucoma.Panretinal photocoagulation (PRP) as well as intravitreal injection of anti-VEGF (bevacizumab) was promptly done. The patient followed up after 1 week with resolution of rubeosis and lowering of IOP back to target levels. After 1 month, repeat ophthalmologic evaluation showed persistence of minimal macular edema, and the patient was given another intravitreal injection of bevacizumab. Subsequent follow-up examinations showed stable eye findings. At the time of the last visit (3 months following treatment), BCVA stabilized at hand motions, anterior segment findings were unremarkable, and IOP maintained at the low teens. Fundoscopy showed widespread retinal atrophic changes with sclerosis of all retinal vessels, as well as optic atrophy Figure 6 . There wThe patient was referred back to his cardiologist for cardiovascular risk assessment, particularly carotid evaluation. Carotid ultrasound with Doppler scan was unremarkable. The patient was advised to continue oral medications for hypertension.Retinal vascular occlusions can occur as a complication of trabeculectomy, particularly in patients with coexisting cardiovascular diseases . It is tCCRAVO is a rare vascular event. The diagnosis is based on ophthalmoscopic fundus findings which combines the features of both CRAO and CRVO, including tortuosity and dilation of the retinal veins, swelling and hyperemia of the optic disc, intraretinal hemorrhages, superficial ischemic retinal whitening with a cherry-red spot, cotton wool spots, or Roth spots . The exaRubeosis iridis develops in 81% of eyes, leading to neovascular glaucoma. This can be seen as early as 1–2 weeks, but on average at about 6 weeks . PreviouThe management of CCRAVO is likewise not well established. In eyes with neovascularization, aggressive treatment with PRP is recommended . HoweverIn the case presented, CCRAVO occurred in an eye that already had a poor visual prognosis due to advanced glaucoma. Prompt treatment with PRP and intravitreal anti-VEGF managed to control the neovascularization and lower IOP, with return to baseline BCVA. However, continuous follow-up and monitoring are still needed in these cases. CCRAVO is usually associated with sequelae such as optic atrophy, neovascularization, and vitreous hemorrhage .This case presented the diagnosis and management of CCRAVO occurring as a complication of glaucoma filtering surgery. While there is literature discussing the mechanisms behind retinal vascular events occurring after glaucoma filtering surgery, it should be noted that the relationship between CCRAVO and trabeculectomy in this case may not be causal and is a limitation of this case report.Retinal vascular occlusions can occur as an uncommon complication of glaucoma filtering surgery, particularly in patients with pre-existing cardiovascular conditions. Combined central retinal artery and vein occlusion (CCRAVO) is a rare retinal vascular condition which combines the features of both central retinal vein occlusion (CRVO) and central retinal artery occlusion (CRAO). CCRAVO can lead to rubeosis iridis and eventually secondary neovascular glaucoma. Prompt panretinal photocoagulation is recommended. Despite aggressive treatment, the visual prognosis in eyes with CCRAVO is usually poor.The authors declare that they have no competing interests.


Extrapulmonary tuberculosis commonly affects the lymphatic system, nervous system, and gastrointestinal system. Tuberculous infection in the muscle is very rare. Moreover, tuberculous infection in the diaphragm is extremely rare. We herein report a case of tuberculomas in the diaphragm and posterior mediastinum that were successfully diagnosed and treated.We encountered a 62-year-old woman with a tuberculoma in the diaphragm. The patient presented with mild dyspnea. Computed tomography showed a mass in the left diaphragm, focal thickening of the posterior mediastinum, and multiple nodules in the lungs. Positron emission tomography–computed tomography showed increased uptake in the left diaphragm mass and thickening of the posterior mediastinum; therefore, we considered the masses to be malignant and planned surgical resection. However, the patient was diagnosed with tuberculosis from a sputum culture, and she was treated with anti-tuberculous therapy. The masses in the diaphragm and posterior mediastinum had become enlarged after 6 months of anti-tuberculous therapy; therefore, the patient underwent resection of both masses. Tuberculous infection was histologically confirmed in each lesion. She was pathologically diagnosed with tuberculous abscesses in the diaphragm and posterior mediastinum and began treatment with anti-tuberculosis drugs.Preoperative diagnosis of a tuberculoma in the diaphragm is usually difficult, and surgical intervention is important for both diagnosis and treatment. Tuberculosis is still a major health concern, although the economic status of the general population and the efficacy of anti-tuberculosis drugs have been improved. The lung is one of the most commonly infected organs in patients with tuberculosis, and extrapulmonary tuberculosis commonly affects the lymphatic system, nervous system, and gastrointestinal system . Tubercumax) = 6.9] . Surgical resection of the masses in the left diaphragm and posterior mediastinum was considered because of concern regarding malignancy. However, a sputum culture showed Mycobacterium tuberculosis; therefore, the patient was treated with anti-tuberculosis therapy (ATT) before surgery. A sputum culture was negative 6 months later. Although treatment for tuberculosis seemed successful, the diaphragmatic mass slightly increased in size and the posterior mediastinal thickness became considerably enlarged. We thus considered that these lesions could be malignant, and the patient was admitted to our hospital for surgery. We did not perform a percutaneous CT-guided needle biopsy of diaphragmatic mass for the diagnosis, because we concerned that a percutaneous biopsy might cause a dissemination of the tuberculosis or the tumor.This case involved a 62-year-old woman with tuberculomas in the diaphragm and posterior mediastinum. She had no relevant medical history and had never smoked. She presented with mild dyspnea. Chest X-ray examination showed small nodules in the bilateral lung fields. Blood test results were normal, including the tumor markers carcinoembryonic antigen and carbohydrate antigen 19–9. Computed tomography (CT) of the chest showed a cystic lesion measuring 30 mm in the left diaphragm , rifampicin (450 mg/day), ethambutol (750 mg/day), and pyrazinamide (1200 mg/day) on postoperative day 7. She was discharged on postoperative day 10 with no complications. At the 6-month follow-up, she was asymptomatic with no evidence of recurrence on CT.Macroscopic examination of the lesion in the left diaphragm showed a 68-mm yellow mass surrounded by a thick wall Fig. a, b. MicWe have herein reported a rare case of a tuberculoma in the diaphragm. Extrapulmonary tuberculosis in the muscles is very rare, with an incidence rate of 0.015% , and onlIncluding the present case, four cases involving treatment of a tuberculoma in the diaphragm have been reported –4. The pExtrapulmonary tuberculosis can be challenging to diagnose on the basis of imaging features . EnhanceA paradoxical reaction, which is defined as a transient clinical or radiological worsening of pre-existing tuberculous lesions or the development of new lesions in patients undergoing ATT, may also make the diagnosis difficult . A paradPossible mechanisms underlying the development of tuberculosis in the chest wall include spread via the lymphatics, spread via the bloodstream, and dissemination from the lung to the thoracic space . Peng etThis report has presented a rare case of a tuberculoma in the diaphragm. The diagnosis of a tuberculoma in the diaphragm is difficult without resection, because symptoms and imaging features of tuberculosis are variable and the development of a paradoxical reaction makes it difficult to assess the worsening of tuberculous lesions. Moreover, conservative treatment is mostly ineffective for tuberculomas in the diaphragm. Therefore, surgical resection of tuberculomas in the diaphragm may be important for both diagnosis and treatment.


Cutaneous leiomyoma is a benign tumor, mainly composed of smooth muscle cells and arising from the arrector pili muscle of hair follicles. The diagnosis of leiomyomas is of paramount importance, as they can often be associated with underlying malignancies  and specific genetic mutations. We report the case of a 27-year-old Caucasian male patient that presented to our attention with a rare segmental and Zoosteriform type II leiomyoma. We performed an analysis of the cutaneous lesions using dermoscopy, reflectance confocal microscopy (RCM) and histology. We found that, using dermoscopy, the leiomyomas showed a dermatofibroma-like appearance with a central hypopigmented area, peripheral delicate hyperpigmentation and also erythematous areas and ectatic vessels. RCM, although not specific, showed groups of hypo-reflective areas distributed in the most superficial papillary dermis, which in histology and immunohistochemistry corresponded to the most superficial protrusions in the papillary dermis of the tumoral bundles. Finally, we discuss the management of patients with multiple leiomyomas and stress the fact that, in the cases of multiple leiomyomas, an annual sonography of the kidneys associated with dermatological and (in women) gynecological consultations are needed to ensure the early identification of an underlying tumor. A genetic consultation to detect an eventual FH mutation is recommended, but since in some cases the FH result may be negative, the above recommended controls remain always of paramount importance. Cutaneous leiomyoma is a benign tumor, mainly composed of smooth muscle cells and arising from the arrector pili muscle of hair follicles . LeiomyoA 27-year-old Caucasian male patient presented to our attention for a 2-year-old history of multiple, painful, scattered papulo-erythematous lesions, distributed at the left upper limb and left mammal areas in a segmental and dermatomal-like distribution a,b.His personal and familial medical histories were negative for cutaneous and systemic diseases. Dermoscopy of the lesions showed a central structureless and hypopigmented area with a mild peripheral pigmentation (dermatofibroma-like pattern), some peripheral arceiformes erythematous areas and scattered, ectatic linear vessels .We decided to perform reflectance confocal microscopy (RCM) in three lesions before performing a cutaneous biopsy. RCM showed a general and unspecific alteration of the epidermis with aggregates of some hyporeflective areas, general disorder of the honeycomb pattern within the upper dermis, the presence of multiple and ectatic vessels (with accelerated blood flow) and some structures characterized by hypo-reflective aggregates that penetrated into the deeper dermis (not visible in RCM) a. The cuAccording to the clinic–pathologic correlation, a final diagnosis of segmental Zosteriform type II leiomyoma was rendered. The patient confirmed that no one in the family presented with these cutaneous lesions and/or systemic malignancies; a complete abdominal ultrasound was negative, and a medical genetic test (for the detection of a FH mutation) was requested.Cutaneous leiomyomas are single and/or multiple papular cutaneous lesions with a red–brownish appearance; when they are multiple, they usually arise in the trunk and extremities ,2,3,4,5.Multiple piloleiomyomas usually occur in the age group of 10–30 years as in our case, presenting with a disseminated or grouped distribution. Grouped leiomyomas are fairly common; however, leiomyomas with a segmental and Zosteriform distribution are rare. Zosteriform leiomyomas can be categorized into two types as follows: (I) Type I is characterized by the presence of a dermatomal distribution; and (II) Type II is characterized by a segmental disposition, superimposed on scattered and isolated lesions. Our case showed a Zosteriform type II distribution because of the occurrence of the multiple clustered lesions in the left upper limb and left chest; in the literature, very few cases have been reported with this type of distribution ,7,8.In our case, dermoscopy revealed the presence of central hypopigmented areas with a peripheral delicate brownish pigmentation; these aspects were also observed by Di Luvio et al., where leiomyomas showed a dermatofibroma-like appearance . HoweverReflectance confocal microscopy (RCM) is a non-invasive diagnostic method that allows the visualization of cutaneous lesions in vivo at a near histologic resolution, providing the visualization of nuclear and cellular morphology of the skin to a depth of approximately 150 to 200 μm depending on the anatomical site . TherefoAlthough, to date, no official screening guidelines for patients with multiple leiomyomas are present as well as for patients with Hereditary leiomyomatosis and renal cell cancer (HLRCC) syndrome, Smit et al. listed practical criteria to ensure an appropriate diagnostic approach ,11. A ge2 laser) are valid therapeutic options to excise the lesions, although not always with satisfactory results. Finally, for painful cutaneous lesions, nifedipine, phenoxybenzamine and gabapentin may be prescribed to reduce the pain.To date, there is no definitive treatment for cutaneous leiomyomas. Surgical excision can be performed in single cutaneous lesions, while for multiple lesions, the treatment is more difficult; in these cases, nitrogen cryotherapy and carbon-dioxide laser  some RCM features may have clinic, dermatoscopy and pathologic counterparts, allowing to improve the diagnosis of these rare lesions. Finally, we stress the fact that in cases of multiple leiomyomas, an annual sonography of the kidneys associated with dermatological and (in women) gynecological consultations are needed to ensure the early identification of an underlying tumor. A genetic consultation to detect an eventual FH mutation is recommended, but since in some cases the FH result may be negative, the above recommended controls remain always of paramount importance.


Hiatal hernia (HH) contents commonly include stomach, transverse colon, small intestine, and spleen but herniation of the pancreas is an extremely rare phenomenon, even rarer when HH is associated with acute pancreatitis.A 56-year-old female with hypertension and gastroesophageal reflux disease presented with abdominal pain, vomiting and chest discomfort evolving for 24 h. Physical examination revealed left-upper quadrant tenderness without guarding. Blood tests showed elevated serum amylase and lipase levels.An abdominal CT scan demonstrated a large type-IV hiatal hernia involving the entire stomach, transverse and right colon, small intestine, duodenum as well as the head, body and the tail of pancreas. The pancreas was enlarged consistent with pancreatitis. Patient clinical status improved with conservative treatment.The stomach is the most common organ to herniate through the diaphragm and pancreatic herniation is extremely rare with only few cases in the literature. Even rarer when associated with acute pancreatitis. This diagnosis is a major diagnostic and therapeutic challenge that has to be evoked in elderly presenting with chest pain and a negative cardiopulmonary evaluation. The ideal treatment is still unclear, however, conservative treatment is the initial management and surgery may be considered in case of recurrent episodes of acute pancreatitis.HH associated with acute pancreatitis is a major diagnostic and therapeutic challenge. Clinicians should consider this rare diagnosis in every case of chest pain with negative cardiopulmonary evaluation. •Intrathoracic herniation of the pancreas is extremely rare.•Clinicians should be mindful of its resulting complications.•Type-IV hiatal hernia may explain certain cases of idiopathic pancreatitis.•The initial management of acute pancreatitis with intrathoracic pancreas is conservative. Her abdominal pain was located in the left upper quadrant. She described the pain as moderate in intensity, of sudden onset and associated with chest discomfort and dyspnea. She had no significant family history and denied any alcohol abuse. Physical examination showed a heart rate of 82 beats/min, a blood pressure of 147/82 mmHg, a body temperature of 37.4 °C, and a respiratory rate of 22/min. Abdominal examination demonstrated left-upper quadrant tenderness without guarding, the bowel sounds could be perceived on the left chest. Cardiovascular, pulmonary and neurological examination was unremarkable.Blood examination results showed elevated serum amylase level at 920 U/L, elevated serum lipase level at 1027.85 UI/L, leukocytosis at 15,300 U/L with 84 % neutrophils.The alanine aminotransferase level was at 367.4 U/L, the aspartate transaminase level at 233.2 U/L, total bilirubin level at 87.41 umol/l and direct bilirubin level at 47.6 umol/l, alkaline phosphatase level at 236 IU/L, and gamma-glutamyl transpeptidase level at 74 IU/L. The renal function was normal.A Given her clinical presentation, CT of the abdomen and serum lipase elevation, a diagnosis of mild acute pancreatitis was made and the patient was treated with intravenous hydration with lactated Ringer's Solution, analgesics along with intensive care monitoring. Etiology investigation of pancreatitis was negative arguing in favor of the hernia as a cause of acute pancreatitis in this case.An esophagogastroduodenoscopy confirmed hiatal hernia without obstruction and excluded the presence of esophagitis. Biopsies revealed chronic gastritis in the antrum of the stomach. We had to respect the declared intention of the patient for a conservative procedure. She was discharged 5 days later. The medical follow up visit two weeks later showed a recovered patient. Upon discussion, we explained that a delayed surgery should be attempted to prevent the increased morbidity and mortality related to a potential recurrent pancreatitis. However, the patient insisted on refusing any surgical intervention.3Hiatal hernias are a part of diaphragmatic hernias Because the head segment of the pancreas and duodenum are positioned in the retroperitoneum and held by Treitz's ligament, pancreatic herniation is extremely rare As a result, pancreatic hiatal hernia associated with acute pancreatitis is extremely unusual, having been recorded in only 7 cases previously Symptoms of acute pancreatitis caused by the herniation of pancreas in HH consist primarily of chest and epigastric pain Cases of pancreatic herniation with pancreatitis are rare; Therefore, the ideal treatment is still unclear Historically, HH cases with pancreatitis were treated with surgery 4HH-associated acute pancreatitis represents a serious diagnostic and therapeutic challenge. Although rare, this diagnosis should be considered in patients with inexplicable pain associated with a large HH, or, in elderly presenting with chest pain and a negative cardiopulmonary evaluation. Conservative treatment is usually indicated and a case-by-case evaluation is the most appropriate management approach.HHhiatal herniaCTcomputerized tomographyNone.An ethical approval was obtained from the Jendouba Regional Hospital Medical Ethics Committee N° CC19Y22.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Conceptualization: AM.Data curation: KA.Supervision and performing surgery: AM.Writing - original draft: MFA, JY.Writing - review & editing: AM.The final version of manuscript was read and approved by all authors.Not applicable.Atef Mejri.Not commissioned, externally peer-reviewed.The authors declare that they have no conflicts of interest.


Sialolithiasis is a common nonneoplastic disease of the major salivary glands that often affects the submandibular glands. Minor salivary gland involvement by sialolithiasis is uncommon, with only 273 cases reported. A long clinical history, acute symptoms, and mucopurulent discharge are unusual features of these cases. Herein, we report the case of a 63-year-old woman who complained of symptomatic nodular swelling of the buccal mucosa associated with purulent discharge for several days. The clinical history lasted 15 years, with episodes of asymptomatic non-suppurative swelling in the same area. The patient underwent surgical excision. The microscopic examination revealed chronic nonspecific sialadenitis associated with psammomatous calcifications, confirming minor salivary gland sialolithiasis. After 3 years of follow-up, the patient was free of symptoms. Patients with sialolithiasis are usually asymptomatic; however, swelling, pain, and fistula may be present in rare cases. The presence of purulent exudate should lead to the differential diagnosis of stomatitis glandularis, a rare inflammatory condition affecting the minor salivary glands. Sialolithiasis and stomatitis glandularis should be considered in the clinical differential diagnosis of symptomatic suppurative nodular swelling affecting the oral mucosa, and histopathological analysis is necessary for the diagnosis. Recurrent or chronic sialadenitis is more likely to be inflammatory, as observed in recurrent childhood parotitis and sialolithiasis. Salivary gland inflammation is caused by salivary duct obstruction due to lithiasis or salivary duct stricture.1-In 1965, Papin1The present case report aimed to describe an unusual case of a patient with SMSG on the buccal mucosa, which exhibited purulent drainage and pain after 15 years of evolution.A 63-year-old woman presented to the oral and maxillofacial department complaining of a painful suppurative lesion on the buccal mucosa for several days. The patient reported several episodes of asymptomatic, non-suppurative swelling in the same lesion area over the last 15 years. The patient’s medical history was unremarkable. Intraoral examination revealed a painful nodular swelling of the buccal mucosa with mucopurulent discharge A. The coDuring surgery, a spherical, whitish calcified mass compatible with sialoliths was depicted A. GrosslThe microscopic examination revealed a fibrovascular stroma containing foci of neutrophil infiltration, acinar atrophy, and numerous dilated salivary gland ducts surrounded by an abundant chronic inflammatory infiltrate A. The ha-Sialolithiasis is a common salivary gland disease that occurs more frequently in middle-aged patients with a slight male predominance. Sialolithiasis commonly affects the submandibular gland (80%), followed by the parotid gland (6%–20%); sublingual and minor salivary gland involvement rarely occurs (2%).1Staphylococcus aureus, mainly affecting the parotid glands.3-The SMSG usually presents as a palpable, submucosal nodule and is often asymptomatic; and mostly involves patients with a mean age of 55 years and men. The upper lip and buccal mucosa are the most affected sites, followed by the lower lip and mucobuccal folds. The signs and symptoms depend on the size and location of the sialoliths. The time of symptoms varies from several months to 3 years, mostly detected within the first year. In some cases, the lesion compression may render a purulent discharge. In the present case, symptomatic nodular swelling with mucopurulent discharge was detected after 15 years. These clinicopathological features are not common in patients with sialolithiasis. They should be distinguished from acute suppurative sialadenitis, a disease caused by bacterial infection, commonly -Treatment of sialolithiasis depends on the symptoms and the size and localization of sialoliths. Although some sialoliths are removed by manipulation of the salivary gland, most patients require surgical excision, as shown in our case. Another treatment option is lithotripsy during sialendoscopy.1-11-Mucoceles, mucous retention cysts, and salivary gland neoplasms were initially considered in the clinical differential diagnosis when assessing nodular swelling of the oral mucosa. After performing a histopathological analysis, the final diagnosis was sialolithiasis.1Although challenging, SMSG should be considered in the clinical differential diagnosis of suppurative nodular swelling affecting the oral mucosa. Suppuration is possible in patients with sialolithiasis of the minor salivary glands, as reported in this clinical case with a 15-year duration.


Allergic fungal rhinosinusitis (AFRS) is counted as the most common form of fungal sinusitis. It is mainly due to the hypersensitivity reaction to fungal infection. Usually, the patients are atopic or immunocompetent. These patients are usually suffering from signs and symptoms of rhinosinusitis. The expanding mass in the disease leads to bony remodeling and the involvement of adjacent structures. When allergic mucin involves the orbit, many complications may occur. This includes diplopia, telecanthus, unilateral proptosis, malar flattening, epiphora, Asthenopia and even visual loss. The diagnosing of AFRS initially requires radiographic imaging, but to confirm the diagnosis, histopathological examination is needed. The treatment of AFRS should be combined with surgical and medical therapy. This case report demonstrates a unique and rare presentation of the non-invasive AFRS with bilateral proptosis which had dramatic improvement and resolution after we managed it with endoscopic sinus surgery, steroids and nasal saline irrigation. Allergic fungal rhinosinusitis (AFRS) is counted as the most common form of fungal sinusitis. It is mainly due to the hypersensitivity reaction to fungal infection.  Usually,A 35-year-old Saudi male presented to our ENT Clinic with a 1-year history of progressive facial pain, headache, bilateral proptosis, anosmia and nasal obstruction. His past medical history was insignificant. Sinonasal endoscopy revealed multiple bilateral grade IV polyps, occluding the nasal cavities bilaterally. Computed tomography (CT) of the paranasal sinuses showed expansion and remodeling of the frontal sinuses bilaterally . It is aThe patient underwent functional endoscopic sinus surgery. The polyps were removed completely from the nasal cavities, bilateral wide maxillary sinus enterostomies, and thick mucin was aspirated. The anterior and posterior ethmoids sinuses were full of polyps, mucin and fungal mud which was aspirated the lamina papyruses that were dehiscent bilaterally by the polyps causing the proptosis of both eyes. The frontal recesses and sinuses were full of polyps, mucin and fungal mud causing erosion of frontal floors and displacing the orbits, and all were cleaned and drained with the help of navigation assisted surgery. The patient had dramatic improvement after surgery, the rhinosinusitis symptoms and the proptosis resolved, and the paranasal sinuses were clear. The patient was discharged with topical corticosteroid and saline irrigations.In patients with AFRS, progressive nasal obstruction and multiple nasal polyps are presented with unilateral distribution most of the time. Usually, these patients are suffering from signs and symptoms of rhinosinusitis lasting months or even years, and they may delay visiting the hospital until they develop complete nasal obstruction, visual disturbances or facial disfiguration . The accThe direction of the least resistance and the location of the disease will determine the site of expansion. The lamina papyracea is the most common location of bone erosion, and the orbit is the most common location for extra-sinus disease spread , 9. WhenLoss of vision can be caused by two mechanisms including, the inflammatory process of adjacent structures that may affect the optic nerve, resulting in optic neuritis or infarction of the optic nerve caused by increased intra-orbital pressure which may lead also to thrombophlebitis within the valveless orbital veins or even occlusion of the central retinal artery which is an ocular emergency , 6, 9. FIn advanced cases, the base of the skull may be involved, leading to an intracranial extension which may give rise to frontal lobe compression leading to change in personality and behavior, or even an intracranial abscess .The diagnosing of AFRS initially requires radiographic imaging. Paranasal sinus CT without contrast is the preferred modality. Its findings usually include irregular hyperdense masses involving multiple sinuses. The ethmoid sinus is most commonly affected, followed by the maxillary, frontal and sphenoid sinuses, respectively. MRI is a good alternative, which may reveal enhanced sinus mucosa and masses with markedly decreased intensities resembling air on T2 images. The radiological findings obtained from CT and MRI are characteristic of dense extra-mucosal mucin, but they are not enough to diagnose AFRS. The definitive diagnosis is reached by histopathological examination of mucosa and mucin , 9, 10. The treatment of AFRS should be combined with surgical and medical therapy, which consists of sinuses surgical decompression, corticosteroid and nasal saline irrigation. Adjuvant medical therapy aims to decrease inflammation, atopy and antigen exposure, therefore, the immunoreactions. In the absence of this combination, the recurrence rates of AFRS reach up to 100%. , 6–9.This case report demonstrates a unique and rare presentation of the non-invasive AFRS with bilateral proptosis which had dramatic improvement and resolution of the proptosis after we managed it with Endoscopic sinus surgery, steroids and nasal saline irrigation.The authors declare that there is no conflict of interest regarding the publication of this paper.There is no financial support and sponsorship.Written informed consent was obtained from the parents for publication of this case report on behalf of the patient.


Teaching point: Mazabraud’s Syndrome defines the association of bone fibrous dysplasia and intramuscular myxoma, sometimes with pathological fractures or deformities in the lower limbs. A 44-year-old man presented to the emergency room with left abdominal pain which required abdominal computed tomography (CT), evidencing left ureteral lithiasis, multiple osteolytic lesions with sclerotic margins on lower ribs, thoracolumbar spine, pelvis and right femur , arrow. As the patient indicated intermittent right gluteal and lumbar pain over the last months without any functional impairment, pelvic magnetic resonance imaging (MRI) was performed for characterization.It showed numerous intra-osseous lesions with hypointense and heterogenous hypersignal respectively on T1-weighted , circle The intramuscular lesion was T1 hypointense and T2 hyperintense  are the most frequent situation .The pathology is caused by inactivating mutations of the GNAS1 gene that induce aberrant cell proliferation at different stages of cellular maturation. Because the physiopathology involves early-stage cells, Mazabraud’s syndrome can present wide clinical forms such as bone pain, deformities or pathological fractures, but patients are sometimes asymptomatic or paucisymptomatic like ours [On radiographs, fibrous dysplasia looks like a radiolucent lesion, appearing in “ground-glass” matrix, with sclerotic bone rim. The bone cortical stays intact .MRI is the gold standard exam to evaluate Mazabraud’s syndrome, for both diagnosis and follow-up with detection of malignant evolution .Fibrous dysplasia appears generally with hyposignal on T1-weighted images and hypersignal on T2 sequence surrounding by hypointense on both T1 and T2 sequences border. There is generally low heterogeneous contrast enhancement. Intramuscular Myxoma shows low intensity on T1 and hyper intensity on T2 [Treatment is variable, from clinical and imaging follow-up to orthopedic surgery for symptomatic patient. Long-term bone antiresorptive agents (bisphosphonates) like zoledronate would be an alternative. It seems reduce the volume of myxoma but doesn’t have any effect on dysplasia .


Caecal volvulus (CV) is a rare cause of intestinal obstruction, defined by an axial torsion of the caecum, ascending colon, and terminal ileum around the mesenteric vascular pedicles, leading to ischemia and bowel necrosis. A 20-year-old woman, with no significant medical history, was admitted for generalized abdominal pain evolving for three days, along with constipation and abdominal distension, but with no vomiting. Physical examination showed a generalized abdominal tenderness with no rigidity or rebound tenderness, associated with abdominal distension and tympanic upon percussion. Laboratory findings were within normal limits. An abdominal computed tomography scan revealed distension of a loop of the large bowel with its long axis extending from the right lower quadrant to the epigastrium or left upper quadrant. Colonic haustral pattern was absent. An abdominal computed tomography scan showed a rounded focal collection of air-distended bowel with haustral creases in the upper left quadrant. In addition, spiraled loops of the collapsed cecum (giving a whirl sign) were noted, along with low-attenuating fatty mesentery from the twisted bowel. The patient underwent an emergency laparotomy and caecectomy using GEA 80 charges. The patient had no complaints post-operation. CV is a rare cause of bowel obstruction, mainly caused by an exceedingly mobile caecum. Despite its rareness, CV represents the second most common cause of large bowel volvulus, behind sigmoid volvulus. For acute obstruction by CV, it is hard to differentiate it clinically from obstruction of the small bowel; therefore, radiological exams are needed. Surgery is the gold standard treatment for CV. We report a rare case of CV to highlight the rarity of this pathology, specify its diagnostic and therapeutic means, and its clinical and biological evolution. Volvulus is commonly defined as a twisted loop of the intestinal bowel and associated mesentery around a fixed point at its base. Caecal volvulus (CV) is a rare cause of intestinal obstruction, defined by an axial torsion of the caecum, ascending colon, and terminal ileum around the mesenteric vascular pedicles.Preoperative diagnosis can be difficult due to its unspecific symptoms. As a result, CV is a surgical emergency and any delay in management can be associated with complications mainly bowel ischemia eventually leading to perforation and peritonitis.We report this case of CV to highlight the rarity of this pathology, the difficulty of clinical diagnosis, and to report the success of caecostomy as a surgical option against right colectomy.A 20-year-old Tunisian woman, unemployed, with no significant medical history, was admitted for generalized abdominal pain evolving for three days, along with constipation and abdominal distension, but with no vomiting. Physical examination showed a generalized abdominal tenderness with no rigidity or rebound tenderness, associated with abdominal distension and tympanic upon percussion. Laboratory findings were within normal limits.Abdominal X-rays were taken, and they revealed distension of a loop of the large bowel, with its long axis extending from the right lower quadrant to the epigastrium or left upper quadrant. Colonic haustral pattern was absent .An abdominal computed tomography (CT) scan showed a rounded focal collection of air-distended bowel with haustral creases in the upper left quadrant. In addition, spiralled loops of collapsed cecum (giving a whirl sign) were noted, with low-attenuating fatty mesentery from the twisted bowel .The patient was kept for six hours under observation, with nothing by mouth, nasogastric tube suction, and IV fluids. At the end of the evaluation period (six hours post suction), the patient underwent an emergency laparotomy.During the operation, the caecum in the left hypochondrium was hugely distended and contained signs of pre-perforation, such as bowel deperitonization lesions . CV was The patient had an adequate postoperative evolution. Antibiotics prophylaxis was given for 48 hours post-operatively with amoxicillin (Ac. Clavulanique intravenously at the dosage of 1gr three times a day). Venous thromboembolism prophylaxis was given using enoxaparin subcutaneously at a dosage of 4000 UI once a day. A liquid diet was initiated 48 h after surgery. The patient had no clinical signs of leakage. On day 5, the patient was discharged. After two weeks, the final histopathological examination was performed, reporting caecal necrosis without underlying lesions.In the postoperative assessment after two months, the patient was found to be tolerating oral intake, was asymptomatic, and did not have any signs of weight loss. It is the second most frequent location of colonic volvulus and accounts for up to 60% of cases according to several studies. To be diagnosed with CV, two conditions must be met: an abnormal mobile caecum associated with the lack of attachment of the mesenterium, caecum, or right colon to the posterior peritoneum–; and a fixed point around which the caecum can twist.CV is an infrequent cause of colon obstruction. It is believed that a mobile caecum is caused by deficient colonic fixation to the peritoneum or colon elongation resulting from over-rotation during embryologic development. Clinical presentation is exceedingly variable, but the most common symptoms are abdominal, accompanied by nausea, vomiting, and abdominal distension.A mobile caecum is an anatomical variant, present in 25% of the general population according to some studies. In recurring intermittent volvulus, patients experience pain in their lower right quadrant associated with abdominal dilation partially relived by gas release, such as the case of our patient., For acute obstruction by caecal volvulus, it is hard to differentiate it clinically from obstruction of the small bowel; therefore, radiological exams are needed. If not treated on time, an acute obstruction may progress into a devastating acute obstruction associated with severe abdominal pain, sepsis, and peritoneal irritation caused by necrosis and intestinal perforation due to obstruction and twisted mesenteric vessels.,In general, CV can be presented in three clinical syndromes: recurring intermittent pain, acute abdominal obstruction, and devastating acute obstruction.In terms of diagnosis, CT is the imaging technique of choice, allowing not only confirmation of the diagnosis, but also ruling out other causes of acute obstruction. Coffee bean, bride beak, and whirl signs are the most common observations identified during CT. If gangrene, necrosis, or perforation are identified, resection is mandatory, and the current method of choice is right colectomy with primary anastomosis or ileostomy depending on perioperative factors. Three main procedures are described in the literature following caecum detorsion with no suspicion regarding its viability: isolated detorsion, caecopexy, and caecostomy. Isolated detorsion without caecopexy or cecostomy is associated with a high risk of recurrence; therefore, it should not be used anymore. Caecopexy is obtained by attaching the right colon to the parietal peritoneum with a recurrence rate of up to 40%. Cecostomy is associated with a higher risk of complications, including caecum gangrene, fistula, and leakage. In this case, our patient was young with signs of caecal pre perforation and not prepared for a right colectomy; as a result, we choose to perform a caecostomy. Compared to caecopexy, caecostomy has a higher rate of morbidity and mortality. As a result, caecopexy is recommended for patients with viable intestines not tolerating right colectomy and those suffering from mobile caecum syndrome.,, This, in conjunction with modern-day laparoscopic evolution, has decreased mortality, and post-operative results have markedly improved.Surgery is the main course of treatment for CV, ranging from simple detorsion to right colectomy.Caecal volvulus is a rare cause of bowel obstruction, mainly caused by an exceedingly mobile caecum. Early diagnosis can be difficult due to its unspecific symptoms. Computed tomography plays a major role in a positive diagnosis. The main course of treatment is surgical, and modalities depend on various factors such as patient status and perioperative findings. Nowadays laparoscopic evolution continues to reduce postoperative morbidity.Written informed consent to publish this case report and any associated images was obtained from the patient.All data underlying the results are available as part of the article and no additional source data are required. The authors did not respond to the issues raised in the previous review nor did they implement any changes in the manuscript. I suggest authors to read my previous comments in order to improve their manuscript.Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?PartlyIs the case presented with sufficient detail to be useful for other practitioners?YesIs sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?PartlyIs the background of the case’s history and progression described in sufficient detail?YesReviewer Expertise:Gastrointestinal SurgeryI confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. Comments to the AuthorsAbstractThe findings of the abdominal X-rays are described as they were from the abdominal CT scanThe correct term is “GIA 80 stapler” instead of “GEA 80 charges”Introduction It is mentioned that the objective of this report is to address the success of caecostomy, but in the case presentation it is stated that the patient underwent caecectomy (resection). Which treatment was perfomed? Please clarifyWe suggest using the sentence: “We report A case of (…)Case PresentationWe suggest using the term: “fasted” instead of “nothing by mouth”The correct term is “GIA 80 stapler” instead of “GEA 80 charges”Please specify which treatment was performed: caecostomy or caecectomy (resection). If it was a resection, was primary anastomosis performed?“Clavulanique” is a French wordIf the diagnosis of CV was already made by abdominal CT scan, why was the patient kept under observation for 6 hours? Why was emergency laparotomy or laparoscopic not performed after diagnosis?Discussion The authors state that CV is an infrequent cause of colon obstruction, but then the mention it is responsible for 60% of colonic volvulus. Is this data correct?We suggest using the sentence: “its use is not currently advocated” instead of “it should not be used anymore?The authors stated they performed a caecostomy. Is this correct? Why not resect the caecum? Please clarify which surgery was performed and justify its choice in the discussion Abbassi et al performed an interesting case report of caecal volvulus. Some issues with this manuscript are detailed below.Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?PartlyIs the case presented with sufficient detail to be useful for other practitioners?YesIs sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?PartlyIs the background of the case’s history and progression described in sufficient detail?YesReviewer Expertise:Gastrointestinal SurgeryI confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. This is an interesting paper which describes a case of cecal volvulus (CV). CV is an uncommon cause of colonic obstruction. Diagnosis is difficult and, if delayed, the results may be intestinal ischemia, perforation, sepsis, and even death. First-line treatment for cecal volvulus is surgery, as nonoperative management is rarely achievable. That Is why I want to ask the authors why they perform an evaluation period of six hours? They must insist that Caecum Volvulus requires immediate surgery because the delay in diagnosis is responsible of high rate of mortality. They should also mention if there was previous abdominal surgery, history of chronic colicky abdominal pain or intermittent bowel subocclusion. Please correct those sentences: “After CV was diagnosed, a caecectomy was performed using GEA 80 charges. -> After CV was diagnosed, a caecectomy was performed using GIA 80 surgical staplers” “A mobile caecum is an anatomical variant, present in 25% of the general population according to some studies." This is overestimated because mobility of the cecum and ascending colon has been estimated to occur in 10–20 % of population. You can mention that several authors have reported successful colonoscopic reduction of cecal volvulus. The manuscript is well described and all the other parts are well described with adequate diagnostic steps.Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?YesIs the case presented with sufficient detail to be useful for other practitioners?YesIs sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?YesIs the background of the case’s history and progression described in sufficient detail?YesReviewer Expertise:surgeryI confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. This was a good case report written in a proper manner.Better to mention in the history that there was no history of previous abdominal operation.Why was the case kept on observation for 6 hours while it is very clear case of intestinal obstruction, only resuscitation for 1-2 hours enough?During the operation it is better to put clamps on proximal and distal before de-rotation to prevent endotoxins being liberated into circulation. Some few comments for the authors in the management of case:Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?YesIs the case presented with sufficient detail to be useful for other practitioners?YesIs sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?YesIs the background of the case’s history and progression described in sufficient detail?YesReviewer Expertise:NAI confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.


Internal hernias through the Foramen of Winslow account for 0.1% of abdominal hernias and 8% of internal hernias, with a mortality rate of 36%-49%. Cecal volvulus accounts for only 1%-1.5% of all intestinal obstructions with a mortality rate of up to 48%. We present a case of a 56-year-old female evaluated for lower abdominal pain and nausea who received a right hemicolectomy after reduction of a cecal volvulus with internal herniation through the Foramen of Winslow. The ambiguous presentation can complicate initial management, but early detection is essential for quick operative repair and prevention of onset or progression of bowel ischemia and necrosis. Internal hernias are not very common, but the high risk of strangulation and ischemia of the herniated bowel can become a surgical emergency and potentially fatal. Thus it is critical to have internal hernias higher on the differential diagnosis . InternaThe patient is a 56-year-old female with a past medical history of anxiety and migraines. She has a past surgical history of laparoscopically assisted vaginal hysterectomy. She presented to a freestanding emergency room (ER) with five hours of constant and severe lower abdominal pain with associated nausea. She denied any previous episodes of similar symptoms. On physical exam in the freestanding ER, her abdomen was soft and nondistended with generalized tenderness. The patient received a CT of the abdomen and pelvis showing dilated small bowel loops concerning for small bowel obstruction. She was then transferred to the hospital, and a repeat CT revealed possible cecal volvulus Figures -2. Upon The patient was taken to surgery for an exploratory laparotomy. During operation, it was found that the patient had a distended, dusky cecum and right colon Figure , which hThe patient had no further complications or significant side effects on post-op day 5 and was discharged home. Tissue pathology of the specimen resulted showing chronic inflammation and superficial mucosal ulceration consistent with clinical diagnosis of cecal volvulus. The appendix was unremarkable and there was no evidence of dysplasia or malignancy. The patient followed up in the clinic two weeks post-op. She reported improving watery stools and mild abdominal soreness but denied fever, chills, diet intolerance, nausea, or vomiting. She overall reported doing well and would return to clinic as needed.Cecal volvulus is caused by twisting of the cecum, along with the terminal ileum and ascending colon, along its own axis . Cecal vCecal volvulus presents with relatively nonspecific signs and symptoms, as seen in this case. At the time of presentation, the patient complained of constant and severe abdominal pain and persistent nausea. There is a vast differential diagnosis for such a presentation, but rarely is cecal volvulus at the top of the list.Risk factors for an internal herniation through the Foramen of Winslow include an abnormally long bowel, enlargement of the Foramen of Winslow, and changes in the intra-abdominal pressure . The patDue to their associated high mortality, cecal volvulus and internal herniation should be considered when evaluating patients with a suspected small bowel obstruction. Additionally, priority should be placed on early CT diagnosis and, if necessary, subsequent exploratory laparotomy to restore blood flow to the bowel or resect potentially ischemic portions of the gastrointestinal (GI) tract.Through the review of literature, there are limited cases available with the unique combination of volvulus and internal herniation. In the documented cases, the ultimate management is exploratory laparotomy with reduction of volvulus from the foramen, and resection of any ischemic bowel . Internal herniation of the right colon through the Foramen of Winslow is a very rare condition with potential severe complications. These patients can present with vague gastrointestinal symptoms, such as lower abdominal pain and nausea. These nonspecific symptoms can complicate initial management, but early detection is important for quick operative repair and prevention of bowel ischemia, necrosis, and perforation.