Bladder Paraganglioma is a rare type of bladder tumor (0.06%). It is typically benign and the most common symptoms are hematuria, hypertension and headache. About 14% of these tumors are malign and consequently radio and chemoresistants. Therefore, surgery is the mainstay of treatment. As they are likely to recur and to metastize lifelong follow-up is required. The authors report a rare case of a 53 years old man with hematuria and a previous history of micturition syncope who was diagnosed with bladder lesion. During the transurethral ressection of bladder he became severely hypertensive. Plasma metanephrines, and urinary vanillylmandelic acid, were still high and the exams suggested residual tumor. The patient underwent radical cistoprostatectomy. After 4 years of follow-up the patient remains disease free. Approximately 10% of pheocromocytomas occur at extra-adrenal sites . These tHistory of a previously healthy 53 years old male patient with a history of 3 episodes of painless gross hematuria, and one micturition syncope. A contrasted-enhanced CT scan (CECT) was performed and there was a contrast enhanced bladder mass with full wall invasion . No lympPheochromocytomas are catecholamine-secreting tumors derived from adrenal glands and the sympathetic nervous system. Extra-adrenal pheochromocytomas, known as paragangliomas, account for about 15% of all pheochromocytomas . Germ liet al., came to the conclusion that metastatic dissemination is the only real proof of malignancy [et al. suggested that post-operative follow-up protocols should include annual cystoscopy, plasma or urine catecholamine analysis and an MIBG scan, while Young et al., regarded anual urine and serum VMA to be the best tools for detecting clinical recurrence or distant metastasis [Histologically there are no definitive characteristics which reliably distinguish benign from malignant tumours. The most well-known pathological finding refers to the “Zellballen” pattern, in which immunohistochemical staining is usually positive for synaptophysin and chromogranin, with S-100 being highlighted in sustentacular cells. A review of studies looking at other factors  for determination of malignancy by Pattarino lignancy . Pre-opelignancy . The inttastasis . Like feBladder paraganglioma is a rare condition and patients can present with various clinical presentations. Young age, extensive local disease and micturition attacks are risk factors for malignancy while features such as vascular invasion, a deeply invasive growth patterns and recurrence are often poor prognostic signs. Like our study shows, malignant phaeochromocytoma remains a challenging entity to diagnose and treat. Surgical extirpation remains the only cure for these patients and further research into this rare condition is warranted. This case highlights one of the clinical presentation of bladder paraganglioma and provides a clinical review of the current literature on management of this rare condition. Due to its rarety we would like to add our experience to the few cases published so far.


Jackstones are stones in the urinary tract that have the characteristic appearance resembling six-pointed toy jacks. They are nearly always reported to occur in the urinary bladder, and the occurrence in less capacious renal pelvis is unusual. We report a solitary, typical jackstone in the renal pelvis without significant outflow obstruction that was successfully treated with retrograde intrarenal surgery followed by extracorporeal shockwave lithotripsy. This highlights the complex pathophysiological mechanisms in stone formation which needs to be further studied. It is important to recognize the characteristic shape of the renal calculi on the radiological investigation in the diagnosis of the jackstones. Jackstones are stones in the urinary tract that have the characteristic appearance resembling six-pointed toy jacks. They are nearly always reported to occur in the urinary bladder , 2. InteA 63-year-old woman presented with painless intermittent visible haematuria of 4 months duration with no history of flank pain, fever, or vomiting. She had type 2 diabetes mellitus for 18 years. Clinical examination was unremarkable. X-ray KUB showed a typical jackstone in the right kidney . NoncontJackstone in the kidney is exceedingly rare, and our review of the literature revealed only few previous reports , 5. GrasOur case is unique as the patient presented with a solitary large jackstone (2.3 × 2.4 cm) that had a typical appearance as that of jackstones found in the bladder. In the previous case report by Grases et al., the typical appearance as seen with bladder jackstones was absent. The possible reason for the typical appearance in our patient may be the capacious renal pelvis that may have facilitated the formation of the jackstone. The composition of the jackstone in our patient is most likely COM. This is because the stone density was rather high (1355 HU), and its surface was relatively smooth . In suchThe exact pathophysiology of development of jackstone is poorly understood. Outflow obstruction remains the most common cause of bladder calculi in adults which was also seen with jackstones in the bladder . HoweverWe report a solitary, large, typical jackstone in the renal pelvis without significant outflow obstruction. This highlights the complex pathophysiological mechanisms in stone formation within the urinary system which needs to be further studied. It is important to recognize the characteristic shape of the renal calculi on the radiological investigation in the diagnosis of the jackstones.


Traumatic left ventricular pseudoaneurysms are rare and surgical correction is the treatment of choice. In this article, it is reported a case of a myocardial stab injury with primary suture and development of a giant pseudoaneurysm, five years later, that underwent surgical repair. Thus, unlikely true aneurysms, a LVP contains no endocardium or myocardium, being more propense to ruptures-4. Trauma is a rare cause, given the high mortality in this scenario.Left ventricular pseudoaneurysms (LVP) form in the presence of a cardiac rupture contained by surrounding structures, like the pericardium or scar tissue.Because patients frequently present with nonspecific symptoms, a high index of suspicion is needed to make the diagnosis. Whereas electrocardiography and chest X-ray abnormalities are almost always present, they are also usually nonspecificThis is the report of the investigation and treatment of a case of penetrating thoracic trauma by stab with initial surgical repair evolving to a giant LVP after five years of indolent evolution.A 34-year-old female, inmate, reported intermittent episodes of exercise-related chest pain and mild dyspnea. The patient reported a traumatic stab injury in the chest five years before that underwent surgery with suture of pulmonary and myocardial lacerations.In the physical examination, patient had blood pressure and heart rate between normal values with a regular two-stroke heart rhythm. Continuous 4+/6+ murmur with fremitus and cervical irradiation were noted.Mediastinal mass with left cardiac silhouette lost and opacification of the retrosternal space is shown .Sinusal rhythm with signs of left ventricular overload .Moderate dilatation of left ventricle (LV) with a large pseudoaneurysm in the mid-anterior wall. Severe ejection fraction impairment (31%) with mid-anterior and apical anterior hypokinesis . BidirecMagnetic resonance imaging (MRI) was able to precisely evaluate the focal disruption of the left ventricular mid-anterior wall , also alLarge pseudoaneurysm cavity with complete obstruction of mid segment of left anterior descending coronary artery at the level of LVP neck .The patient underwent surgical correction of the pseudoaneurysm by direct suture with good evolution. Medium sternotomy was performed after femoro-femoral cardiopulmonary bypass (CPB) had been established. The patient was cooled down to 28ºC. The ascending aorta was not cross-clamped. The fibrous wall of the pseudoaneurysm was identified and its medial surface (contiguous to the anterior mediastinum) was widely opened during a short period of circulatory arrest and ventricular fibrillation, in order to depressurize the LV and the pseudoaneurysm and to prevent air embolism. The left ventricular wound was identified (3.5 cm ´ 5 cm) and the CPB restarted. The wound was closed using a bovine pericardial patch sutured to its margin with horizontal mattress stiches  during ventricular fibrillation. Maneuvers to remove air from the LV were performed before tying the last stich. Besides a mediastinal drain, a suction drain was positioned inside of the pseudoaneurysm cavity (left open to the anterior mediastinum) and the chest was closed as usual. The suction drain was removed few days later, when the serosanguineous drainage was < 100 ml/24 h.Another cardiac MRI was made six months latter showing a minimal residual cavity without signs of complication. After surgery, the patient had a good postoperative evolution without complications, being discharged from hospital after 11 days. She is at clinical and imaging follow-up, with a two-year evolution, without worsening of symptoms.,7,8.The incidence of LVP is low and most of the cases are related to acute myocardial infarction, cardiac surgery, and trauma. LVP have become a rare complication of acute myocardial infarction, occurring in approximately 2% of the cases or less, when thrombolytic or primary percutaneous intervention can be performed and data to guide the treatment are scarce. Surgical repair is the recommended treatment, given the pseudoaneurysms’ propensity for life-threatening complications, such as cardiac tamponade and shock. Untreated pseudoaneurysms have a 30% to 45% risk of rupture and the surgical repair of post-traumatic LVP has a reported mortality of > 7%.The natural history of surgically treated and untreated LVP is not clearly definedIs this article, it is reported an unusual case of post-traumatic giant LVP with an indolent evolution, although all the potential risks of rupture.


Posttraumatic carotid-cavernous fistula (CCF) is a very rare complication that can occur in patients with craniomaxillofacial trauma. It is defined by abnormal communication between arteries and veins located in the cavernous sinus. CCFs can be divided into two groups: direct, which are usually post traumatic and classically with a high flow and acute onset of symptoms. On the other hand, indirect CCFs are in the most of cases idiopathic and typically insidious of onset. The aim of the present case report is to describe an atypical presentation of direct CCF characterized by the insidious onset of symptoms with the goal to think about this rare complication and so not to delay the treatment which is an emergency in this case. CCF is a very rare complication that can occur in patients with craniomaxillofacial trauma . It is dWe report the case of 27 year-old man who suffered left cranio-facial trauma, caused by motorcycle accident. Consequently the patient presented mandibular fracture which was fixed. four months after the accident, the patient consult for left red eye, sensation of progressive proptosis and the hearing of buzzing sound with progressive intensity, without pain nor visual impairment. The ophthalmologic examination was notable in the left eye for axil, reducible exophthalmos with thrill on palpation , and dilAfter such clinical findings, the diagnosis of CCF was the most probable. We completed with head arteriography. As expected this test revealed the presence of left CCF with shunt onthe C3-C4 segment of the internal carotid artery (ICA).The venous drainage was via posterior into the inferior petrosal sinus, lateral into the sphenoparietal sinus and the superior sagittal sinus, inside into the contralateral cavernous sinus and anterior the thrombosed superior ophthalmic vein was slightly opacified . EndovasCCFs are rare trauma complication . IncidenDirect CCFs are usually post head trauma, secondary to motor vehicle accidents or penetrating injuries, they may also be caused by rupture of intra-cavernous aneurysm or head surgery ,5,6. OurThe main symptoms of CCFs include orbital bruit which can be heard by the patient and objectively determined by the physician, proptosis, chemosis , arterialization of the episcleral veins with the typical corkscrew-like aspect, eyelid edema, diplopia, visual impairment with different degrees and elevated intra ocular pressure. The ophthalmologic examination may find also dilation of retinal veins, intraretinal hemorrhages, mild optic disc swelling and even non-rhegmatogenous retinal detachments and choroidal detachments . Our patDiagnosis is based on the angiographic study, this exam allows the correct lassification of each case and also determine the treatment strategy . In caseThroughout our case report we described an atypical presentation of direct CCF characterized by the insidious onset of symptoms with the goal to think about this rare complication and so not to delay the treatment which is an emergency in this case.The authors declare no competing interests.


Creutzfeldt-Jakob disease (CJD) is the most common human prion disease presenting with subacute cognitive decline. Common MRI findings for CJD include the T2 prolongation signal of the putamen and head of caudate. Diffusion-weighted MRI (DW-MRI) is considered to be the most sensitive technique for the detection of CJD-related abnormalities, especially for cortical changes. We report the case of a 77-year-old female who presented with dizziness, visual hallucination, and a rapid decline in her mental state shortly after a right knee surgery. Brain MRI with contrast showed cortical and subcortical T2 fluid-attenuated inversion recovery (FLAIR) hyperintensities in bilateral posterior temporal lobes and the left occipital lobe without an associated enhancement, suggestive of posterior reversible encephalopathy syndrome (PRES). Workup including metabolic, infectious, and vasculitic panels were all within normal limits. A few days later, she developed persistent myoclonus, and a continuous electroencephalogram (EEG) revealed multifocal epileptiform and generalized discharges, forming multifocal periodic discharges and generalized periodic discharges (GPDs). Cerebrospinal fluid (CSF) analysis was positive for 14-3-3 and elevated T-tau protein consistent with a diagnosis of sporadic Creutzfeldt-Jakob disease (sCJD). This is a rare case of CJD presenting with a brain MRI resembling PRES. CJD may have various features on MRI, and a high degree of suspicion is required to confirm the diagnosis. Creutzfeldt-Jakob disease (CJD) is a subset of a larger family of fatal neurodegenerative disorders known as transmissible spongiform encephalopathies; it was first described in the 1920s by Creutzfeldt and Jakob [The Centers for Disease Control and Prevention (CDC) has outlined two criteria for probable sCJD diagnosis. This includes a neuropsychiatric disorder plus a positive real-time quaking-induced conversion (RT-QuIC) in cerebrospinal fluid (CSF) or other tissue . AlternaPosterior reversible encephalopathy syndrome (PRES) is a clinic-radiological syndrome characterized by reversible changes occurring within the central nervous system that can be viewed by MRI or CT of the brain . PRES clHere we report an atypical presentation of CJD with brain MRI findings typically associated with PRES.A 77-year-old female with a history of chronic alcohol abuse presented with dizziness and confusion one month after a right knee replacement for degenerative joint disease. She described her dizziness as “lightheadedness” that was not associated with any specific maneuver or changes in position. Prior to undergoing knee surgery, she had been healthy, independent, and abstinent from alcohol. With the onset of the confusion, her family had found her to be “jittery” with sleep disturbances and difficulty concentrating. On examination, she was found to have impaired orientation to time and slow thought processing. Her Mini-Mental State Exam (MMSE) was 21/30, consistent with mild cognitive impairment, and the remainder of her systemic exam was unremarkable. A noncontrast CT of the head was negative for an acute intracranial process, and a follow-up MRI of the brain without contrast was unrevealing for underlying abnormality  revealed multifocal epileptiform and generalized discharges, forming multifocal periodic discharges (MfPDs), and generalized periodic discharges (GPDs) at 0.5-1 Hz suggestive of CJD Figure . A repeaTwenty-five days after the admission, she became increasingly somnolent and less responsive and was subsequently intubated. She was then transferred to the neurointensive care unit (NICU) where she remained intubated and sedated for the duration of the hospital course. She continued to decompensate and the family requested to withdraw care. Previous CSF results had returned positive for 14-3-3 and an elevated t-tau protein consistent with the diagnosis of sCJD. Autopsy and brain biopsy were not performed.Our case demonstrates an atypical presentation of CJD with features of PRES on MRI. Of all available neuroimaging modalities, MRI is the most helpful method in the diagnosis of CJD . Recent While there has been an increase in the use of MRI for diagnosing CJD, atypical findings can be noted in the early clinical stages of sCJD . InteresCommon risk factors for the development of PRES include arterial hypertension, eclampsia or preeclampsia, chemotherapy regimens, organ transplants that require immunosuppressant medications, severe infection, chronic renal failure, and autoimmune diseases . In our In rare cases, PRES can present with edema involving the cerebellum, basal ganglia, brain stem, and more anterior structures such as the frontal lobe . Due to https://link.springer.com/article/10.1007%2Fs12028-018-0606-9).This work has been presented as an abstract at the Neurocritical Care Society (NCS) 16th Annual Meeting (CJD is a subset of a larger family of fatal neurodegenerative disorders known as transmissible spongiform encephalopathies. We reported an atypical presentation of CJD with brain MRI features resembling PRES. CJD may have variable MRI features and a high degree of suspicion is required to establish the diagnosis.


Isolated macrodactyly in adults caused by mosaic pattern PIK3CA mutation can result in significant functional impairment and psychological burden. Due to the rarity of this condition there are no clear treatment guidelines, and those few available are focused on paediatric cases. Reports on surgical management of isolated macrodactyly in adults are lacking. We present here the surgical management through partial amputation of enlarged rays of the right hand in an individual affected by low-grade mosaic PIK3CA mutation. We present a case of surgical management for enlargement of the thumb and index rami (1st & 2nd) of the right hand in a 55-year-old female patient with a low-grade mosaic pattern PIK3CA mutation. The challenge in advanced cases such as this is to maintain the stability of the wrist and hand while preserving a maximum of mobility and function.A 53-year-old female patient presented to our centre for consultation due to progressive isolated gigantism of the right hand paired with pain and loss of function. She had no family history of genetic or congenital malformations. Enlargement of right-hand thumb, index and middle  digits had been noticed at 6 months of age, with progressive growth leading to functional disability.At first presentation to our centre, she had previously undergone eight surgical debulking procedures of the enlarged digits throughout her lifetime. Medical treatment with growth-inhibitors was also attempted yet ineffective. The patient had initially been diagnosed with Proteus syndrome based on clinical presentation. Due to uncertainty of this diagnosis and patient's request, genetic testing was conducted through biopsies from enlarged tissues which confirmed a low-grade mosaic mutation in the PIK3CAgene.Symptoms experienced at this stage were pain and loss of function in the right hand with lack of proper grip function. Physical examination showed tissue enlargement, predominantly around the right wrist and volar aspect of the thumb. The wrist was furthermore hyperextended with the thumb and index digits in a fixated position.Having been informed of risks of surgery, the patient initially selected an expectative approach. However, due to increasing impairment and vascular compromise, the patient ultimately chose to undergo surgical intervention.Supplementary Digital Materials (SDM) 1 & 2 showcase preoperative external and radiographic view of the affected area. The procedure went as follows:As debulking was not possible at this stage, we performed a partial amputation of the enlarged tissues along with wrist stabilization through the superficial and deep digital flexor tendons to the index digit . Incisions were planned to conserve as much skin as possible, mainly on the dorsal aspect.The median nerve was identified and showed extreme enlargement and ulnar deviation caused by a radial exostosis. Numerous (>15) large and rigid branches to the thenar were encountered and severed during further dissection. Branches to the middle and ring rami were spared and showed to normalize along their distal course. However, at wrist level the entire median nerve showed to be hypertrophic.A skin flap on the ulnar side of the proximal index digit was spared including the neurovascular bundle containing a hypertrophic collateral nerve, which was removed to minimize recurrence risk.Thenar muscles also showed hypertrophy, whereas flexor pollicis longus (FPL) and both the FDS2 and FDP2 flexor tendons appeared normally sized. The tendons were further dissected and severed at the level of their insertion. The median nerve normalized in size during its’ proximal course at the proximal wrist crease.The amputation followed in which the thumb and index rami, Trapezium, Trapezoid and Scaphoid bones (STT) were all removed. An exostosis on the palmar side of the Capitate bone was also removed, as well as a large exostosis on the palmar Radius. The Lunate bone luxated easily out of the Lunate facet on the Radius while stability of the third metacarpal-capitate-lunate joints appeared to be preserved. A canal was drilled through Radius, Capitate and base of the middle (3rd) metacarpal (MC3) through which the FDS2 was passed, after which the FDP2 was guided around the MC3 to serve as a static wrist stabilizer. The tendons were sutured onto the FPL. This was a tailored solution for the radiocarpal instability that occurred after resection of the STT bones, combined with the resection of the palmar part of radius including the palmar capitate. This equals resection of the complex with the radio-carpal ligaments attached and loss of the radiolunate, radioscapholunate, radioscaphocapitate and radial collateral ligaments. This resulted in gross instability of the wrist with a spontaneous radiodorsal luxation of the lunate and remnant of the radiocarpal joint. The transosseus ligament reconstruction secured a functionally mobile and stable wrist as can be seen in the video. The spared dorsal skin flap was used for wound closure through staples and ethilon sutures. The technique for wrist stabilisation has been depicted schematically SDM 3–7). Follow up X-ray photographs taken two and three years post-operatively (SDM 8 & 9) showed unchanged position of the MC3-Capitate-Radial alignment compared to immediate post-operative X-ray (SDM 10), and clinical stability was unchanged at two- and three-year postoperative examinations Postoperative treatment consisted of temporary splinting and physiotherapy. Besides phantom pain postoperative course was unremarkable during the two year follow up. Videos 1 and 2  showcase the postoperative function at this stage. The patient preserved excellent function of her right hand. Photographs taken at three years post-operatively depict range of motion and aesthetic result  which is caused by mosaic mutations in the AKT1 gene, PIK3CA-related overgrowth spectrum (PROS) which is caused by mutations in the PIK3CA oncogene, and PTEN hamartoma tumour syndrome (PHTS): caused by somatic PTEN tumour suppressor gene mutations.As clinical manifestations often overlap, genetic testing is crucial in accurate diagnosis of individuals suffering from these overgrowth syndromes. Accurate genetic diagnosis is of clinical importance for non-surgical medical management with targeted growth inhibiting drugs and thus plays a role in therapeutic intervention prior to surgery.,The condition is represented by benign overgrowth of osseous, adipose, nervous and/or cutaneous tissue causing disproportionate enlargement and loss of function of the affected area. The hypertrophy is typically found within an area of nerve distribution with the nerve in question also being enlarged and/or elongated.Due to the rarity of the condition, there are no clear guidelines for surgical management of progressive and isolated macrodactyly. There are a handful of publications which describe surgical management and therapeutic consideration for macrodactyly.Possible interventions described in these reports are surgical debulking of enlarged soft tissue, osteotomies, and complete amputation of affected area have been described for macrodactyly in infants and children.Our case describes successful management and gain of function after partial amputation of progressive macrodactyly in an adult patient who had previously undergone several surgical interventions. Although this can be psychologically challenging in an older patient, partial amputation of the wrist risks resulting in gross instability. Tailored solutions can bring a functionally acceptable result for adult PIK3CA patients with progressive and functionally impairing macrodactyly.


Müllerian abnormalities are present in 0.17% of fertile women and 3.5% of infertile women, and a unicornuate uterus is observed in 0.4% of women. The uterus is normally formed during embryogenesis by the fusion of the two Müllerian ducts. If one of the ducts does not develop, only one Müllerian duct contributes to the uterine development. We report a case of Gravida II, abortion I referred from a primary hospital with a referral paper and sonography stating she had IUFD. She had regular antenatal care follow-up at the primary hospital and had 8 months of amenorrhea. Our ultrasound assessment confirmed the intrauterine fetal demise, but the rudimentary horn pregnancy was missed. Repeated attempts at the induction of labor were tried but unsuccessful. The diagnosis was confirmed at laparotomy. She underwent cesarean section with right intact rudimentary horn removal. A nonviable male fetus with birth weight of 1.2 kg was delivered. Women with this abnormality are asymptomatic and unaware of having a unicornuate uterus. Abdominal pain is the most common presenting symptom with the rudimentary horn, but communicating horn pregnancy is generally asymptomatic in early pregnancy. Early awareness of this rare clinical condition is so crucial especially in developing countries where the availability of new technologies is scarce to explore uterine abnormalities. The patient had uneventful postoperative recovery and was discharged after 3 postoperative days. Congenital malformations of the female genital tract are defined as deviations from normal anatomy resulting from embryological maldevelopment of the Müllerian or paramesonephric ducts. They represent a rather common benign condition with a prevalence of 4–7% .The incidence of pregnancy occurring in a rudimentary horn of the unicornuate uterus is very rare ranging from 1 in 75,000 to 150,000 pregnancies .As most of the rudimentary horns are asymptomatic, only 8% of rudimentary horn pregnancies are diagnosed before the symptoms appear .Expert ultrasonography can reveal some Müllerian anomalies, but confirmation requires magnetic resonance imaging .th and 20th week of gestation, although a rupture has been reported at 34 weeks [Cases with rudimentary horn pregnancy usually result in the rupture of the horn in the second or third trimester, typically between the 1034 weeks .This is a case demonstrating a communicating rudimentary horn connected to a unicornuate uterus, where the diagnosis was revealed after surgery.A 22-year-old Gravida II, abortion I came to our institution on referral from a primary hospital with a referral paper and sonography showing that she had IUFD. She had a spontaneous abortion for which dilation and curettage (D&C) was performed at the primary hospital 16 months back. She had no remarkable medical and surgical histories. She had regular antenatal care follow-up at the primary hospital and had 8 months of amenorrhea.Upon admission, the patient was clinically asymptomatic and stable, showing no uterine contractions and no cervical dilatation. On abdominal examination, the uterus was enlarged equivalent in size to 30 weeks of pregnancy.On examination, her BP was 120/75 mm of Hg, pulse 85/minute, temperature 36.5°C, and respiratory rate 22/minute. FHS was absentObstetric ultrasonography revealed a singleton, intrauterine fetal demise with a gestational age of 32 weeks, estimated fetal weight of 1.3 kg, and negative fetal heartbeat. The placenta was fundal and anteriorly. The cervix was closed, uneffaced, and posterior. All laboratory investigations were normal.th dose. Then, she was given an infusion of normal saline 0.9% with 5 IU of oxytocin, followed by increasing concentrations of the same infusion. Just then, there was no cervical dilation, despite the onset of contractions.Initially, she was administered 50 mcg of vaginal misoprostol at 6-hour intervals, without response after the 6Cervical effacement with no dilatation was seen. Finally, a Foley catheter was used. All attempts were unsuccessful. The patient was counselled and underwent a cesarean section horizontally under spinal anaesthesia.The intraoperative finding was intact rudimentary horn pregnancy. Clamps were applied over the fibrous band on the medial side and the fallopian tube and ovarian and round ligaments on the lateral side; the accessory horn was excised with the dead fetus; and right salpingo-oophorectomy was performed Figures  and 2.On releasing the forceps applied to the fibrous band and probing, a canal was detected between the rudimentary horn and the uterine cavity at the level of the isthmus. The round ligament, ovarian ligament, and tube were connected to the right cornua of the uterus.The patient was advised to follow up. The patient had uneventful postoperative recovery and was discharged after 3 postoperative days.A unicornuate uterus with a rudimentary horn is a congenital uterine anomaly which results from the developmental arrest of the two Müllerian ducts with the simultaneous incomplete fusion of the opposite side .The unicornuate uterus with rudimentary horn may have a cavity that is either in communication with (type A1a) or sealed off from (type A1b) the primary uterine cavity, or it may have failed to canalize entirely and is without a cavity .When a rudimentary horn is present, it is the noncavitary type in 33% of cases, the cavitary but noncommunicating type in 22% of cases, and the cavitary and communicating type in 10% of cases .Most cases of rudimentary horn pregnancy (RHP) provide a diagnostic challenge and are diagnosed after rupture, which leads to emergency surgery, blood transfusions, and increased morbidity .Diagnosis is challenging in this malformation due to a limited field of sonographic view compared to other diagnostic imaging modalities and lateral deviation of the rudimentary horn .Sonographic sensitivity is only 26%, and as the pregnancy advances, the specificity goes down .The common misdiagnosis on ultrasound includes a bicornuate uterus with pregnancy in one horn, uterus didelphys, abdominal pregnancy, or even normal intrauterine pregnancy with an adnexal mass undergoing torsion .It is shown that 62% of the rudimentary horn is preferentially placed on the right, which was revealed in our case report because the left Müller's canal progresses more caudally than the right .Our patient had no renal abnormalities despite approximately 38% of patients having coexisting renal abnormalities. Unilateral renal agenesis is most commonly found; this is always ipsilateral with the rudimentary horn .The literature presently supports prompt excision of the horn, pregnancy, and ipsilateral once a rudimentary horn pregnancy has been diagnosed .Early awareness of this rare clinical condition is so crucial especially in developing countries where the availability of new technologies is scarce to explore uterine abnormalities. A high index of suspicion should be made if there is no response to repeated failed attempts at the induction of labor.


Diffuse large B‐cell lymphoma often presents with extranodal manifestations involving the musculoskeletal system. Shoulder pain is particularly worrisome for malignancy. Individuals presenting with refractory upper extremity complaints should undergo a prompt and thorough evaluation for cancer, as a delay in diagnosis can result in an unfavorable outcome. Diffuse large B‐cell lymphoma often presents with extranodal manifestations involving the musculoskeletal system. Shoulder pain is particularly worrisome for malignancy. Individuals presenting with refractory upper extremity complaints should undergo a prompt and thorough evaluation for cancer, as a delay in diagnosis can result in an unfavorable outcome. In addition, we review the characteristics of other patients with DLBCL mimicking shoulder pathologies and discuss the differential diagnosis and treatment options for this common hematologic malignancy.2A healthy 51‐year‐old man presented to the emergency department with progressively worsening left shoulder pain of four‐month duration that had been acutely exacerbated three days earlier by a ground‐level fall. The patient reported that he first noticed a dull ache when playing golf. Radiographs obtained in the emergency department showed moderate degenerative changes but no acute abnormalities. A diagnosis of rotator cuff tendinitis was established. The patient was subsequently provided with an arm sling and discharged for outpatient follow‐up.The patient was evaluated in clinic approximately three weeks after his initial presentation in the emergency department. He reported minimal pain relief despite daily use of the arm sling and oral naproxen sodium. Physical examination revealed significant tenderness to palpation over the left glenohumeral joint. Range of motion of the left shoulder was restricted with abduction and extension. The Hawkins‐Kennedy test and external rotation lag sign were positive. A presumptive diagnosis of rotator cuff tendinopathy vs tear was established. The patient was referred for magnetic resonance imaging (MRI) and outpatient physical therapy. However, a trial of physical therapy was required for health insurance authorization of imaging; the first session was scheduled for one week after the clinic visit.The patient began physical therapy as planned. However, three days after his first session, he presented to the emergency department complaining of testicular swelling. An ultrasound was performed and revealed hydrocele. The patient was diagnosed with epididymitis and prescribed oral ciprofloxacin. The swelling failed to resolve after a two‐week course of antibiotics. He was subsequently evaluated by urology, who recommended continuing ciprofloxacin for eight weeks. Symptoms had partially resolved following two months of treatment.The left shoulder pain failed to improve after three months and twelve physical therapy sessions. An MRI without contrast was ultimately obtained and revealed severe tendinopathy of the supraspinatus tendon as well as a large tumor replacing the marrow of the proximal humerus Figure . Extensi2), cyclophosphamide (750 mg/m2), doxorubicin (50 mg/m2), vincristine (1.4 mg/m2), and prednisone (R‐CHOP) every three weeks. There were no risk factors for central nervous system disease, as testicular involvement was excluded, and thus, central nervous system‐directed prophylaxis was not administered.CT scans of the neck, chest, abdomen, and pelvis were subsequently acquired. Notable findings included splenomegaly, an enlarged right submandibular lymph node Figure A, a lytiThe patient tolerated the therapy well. After the first cycle of R‐CHOP, the patient reported that his shoulder pain was modestly improved and that his scrotal swelling had resolved. Repeat CT scans were obtained after ten weeks and three cycles of R‐CHOP. Lymphadenopathy was markedly improved in the submandibular Figure B and ret3Diffuse large B‐cell lymphoma is one of the most common hematologic malignancies in the United States, with an annual incidence of approximately 5.6 cases per 100 000 individuals. Historically, the prognosis of DLBCL was relatively poor; prior to the year 2000, the five‐year survival rate ranged from 40% to 50%. However, the approval of rituximab—a core component of the modern DLBCL treatment regimen—by the Food and Drug Administration (FDA) in 1997 has resulted in significantly improved survival. Indeed, five‐year survival increased to 60% in 2002 and has continued to climb steadily over the past two decades.DLBCL most commonly presents in middle age, with a median age of diagnosis of 66 years.Musculoskeletal complaints represent a rare but important presentation of an underlying malignancy. Interestingly, there is a particularly strong correlation between shoulder pain and cancer. A recent study by Pedersen et al demonstrated that a new diagnosis of cancer is established in approximately 1 out of every 142 patients presenting with an initial complaint of adhesive capsulitis (“frozen shoulder”).Malignant lymphoma of bone accounts for 7% of all bone malignancies and <5% of extranodal lymphomas.There are no truly distinctive or pathognomonic signs to reliably differentiate osseous lymphoma from primary bone tumors based on plain film radiography alone, and thus, other imaging modalities are often required to establish a diagnosis. MRI is particularly useful, as it has a high sensitivity for marrow replacement. On T1‐ and T2‐weighted sequences, DLBCL demonstrates hypo‐ and hyperintensity, respectively. However, if there is fibrosis within the tumor, T2‐weighted images may appear hypointense. Contrast‐enhanced CT (CECT) scan can also show marrow infiltration, but offers superior visualization of cortical and trabecular destruction. CECT be used for studying marrow disease and for planning a biopsy when MRI is contraindicated.A definitive diagnosis of DLBCL is established via biopsy. Histologically, DLBCL has a diffuse growth pattern with large cells that are usually five times the size of normal lymphocytes. These cells may resemble immunoblasts—characterized by amphophilic cytoplasm with one eccentric nucleus—or centroblasts with pale or basophilic cytoplasm; chromatin margination that makes the chromatin appear vesicular; and two to three nucleoli near the membrane.The classification of DLBCL is currently evolving due to increasing throughput capacity to analyze genetic material. At present, DLBCL is divided into two broad categories: double/triple‐hit lymphomas (DHLs) with specific cytogenetic rearrangements and nondouble/triple‐hit lymphomas. Nondouble/triple‐hit lymphomas are further subtyped based on the cell of origin into germinal center B‐cell‐like (GCB) and activated B‐cell‐like (ABC) tumors. The specific subtype has important clinical implications, as individuals with GCB tumors typically exhibit superior survival.Our patient presented with shoulder pain that was initially diagnosed as a rotator cuff injury; DLBCL was not identified until several months after his symptoms began. Indeed, although this presentation is rare, other authors have described similar cases. In 2018, Liu et al described a 64‐year‐old woman who presented with six months of worsening right shoulder pain and limited range of motion.A similar case was reported several years earlier by Caporale et alThe individuals described in the above‐referenced cases share several similarities with our patient: the clinical presentation of shoulder pain and limited range of motion; normal findings on plain film radiography; and the absence of constitutional symptoms, such as night sweats, fevers, and weight loss. All three patients were treated with R‐CHOP, which is the current standard of therapy. However, the patient described by Caporale also had surgical excision of the tumor and a reverse shoulder prosthesis due to the size of the mass. In contrast, the patient described by Liu et al exhibited a complete response to chemotherapy alone; a two‐year follow‐up showed increased range of motion, decreased pain, and no evidence of recurrent or metastatic disease.Several other investigators have also described DLBCL presenting as shoulder pain. Characteristics of these patients are summarized in Table TNFAIP3, which encodes a negative regulator of NF‐kB. Indeed, 30% of individuals who develop the ABC subtype are found to have two mutated copies of this gene. The ABC subtype of DLBCL can also be distinguished from the GCB subtype based on the BCL‐6 gene. BCL‐6 is required for plasma cell differentiation, and translocations in this gene are twice as common in the ABC as compared to GCB subtype.The considerable genetic diversity of DLBCL can be attributed to the cells of origin: Antigen‐exposed B cells found within germinal centers. These cells are exposed to an environment that promotes rapid proliferation, cytidine deamination (for receptor editing), and somatic hypermutation.The mainstay of treatment for DLBCL for the past twenty years has been R‐CHOP, which represents the first‐line therapy per the National Comprehensive Cancer Network (NCCN) guidelines. However, the medical community is currently in the midst of a paradigm shift in the management of this common hematologic malignancy, as recently identified cytogenetic markers will allow clinicians to individualize therapies based on specific tumor characteristics. In one recent open‐label randomized trial, investigators found that individuals with the ABC subtype of DLBCL who were treated with rituximab, doxorubicin, cyclophosphamide, vindesine, bleomycin, and prednisone (R‐ACVBP) exhibited improved survival as compared to those who received R‐CHOP. Notably, there was no significant difference in survival among those with the GCB subtype.4Diffuse large B‐cell lymphoma is one of the most common hematologic malignancies in the United States. The classic presenting feature is local or generalized lymphadenopathy. However, extranodal manifestations are common and may involve the central nervous system, gastrointestinal tract, or, as in our patient, the musculoskeletal and genitourinary systems. Shoulder pain and stiffness are particularly worrisome for malignancy. A careful physical examination with appropriate follow‐up laboratory and imaging studies can help establish an early diagnosis. Treatment typically involves chemotherapy, which can be individualized based on DLBCL subtype.None declared.BDB: wrote the case report and edited and finalized the manuscript. RK: wrote and edited the portions of the manuscript on imaging and pathology. PP: wrote and edited the literature review and treatment section of the manuscript. MBU: conceived of the project and helped with language editing. CLE: involved in the case and provided technical editing. JF: involved in the case and provided technical editing. RI: proofread the final draft of the manuscript. NG: supervised the project from initiation to completion.


Background: Colosalpingeal fistula is a rare complication secondary to diverticular disease. The pathogenesis is still not clearly understood. We present the case of a colosalpingeal fistula and a review of the management of this pathology. Case report: A 69-year-old patient with uncomplicated diverticular disease was referred to our department for recurrent vaginal discharge. The clinical examination was unremarkable, hysteroscopy revealed the presence of air in the uterine cavity in the absence of a uterine fistula. A preliminary diagnosis of colosalpingeal fistula was made and was confirmed by computed tomography (CT) scan and hysterosalpingography. A one-stage surgery via laparotomy was successfully performed with remission of the symptoms. Conclusion: Colotubal fistula is a rare complication resulting from intestinal diverticular disease. The purpose of this paper was to emphasize the presence of a rare, but serious complication occurring in diverticular disease with atypical symptoms and one-stage surgery treatment. The colotubal  fistula occurs in 2% of the cases with fistulas secondary to diverticular disease . The iniEscherichia coli and Streptococcus constellatus. The transvaginal ultrasound examination showed a heterogeneous intrauterine collection. Hysteroscopy was performed as an additional exam. The presence of air in the uterine cavity in the absence of uterine fistula or neoplasic process made us suspect the tubo-intestinal fistula. The endometrial biopsy was compatible with endometritis secondary to E. coli. To confirm the diagnosis, CT scan and hysterosalpingography were performed and the communication between the tube and sigmoidal diverticulum was visualized  is an ineffective therapy and it is associated with a higher recurrence risk . This maThe novelty of this case report is that, unlike the literature reports, our patient presented with gynecological problems and her only symptom was the vaginal discharge, managed in our gynecology department.Colosalpingeal fistula is a rare complication of intestinal diverticulosis, and the diagnosis should be suspected by the gynecologist in a patient with abdominal pain and known diverticular disease, especially in the presence of persistent or recurrent vaginal discharge. The purpose of our paper is to bring attention to the recurrent vaginal discharge and to make the differential diagnosis with the colotubal fistula, especially in patients with diverticular or inflammatory disease.


Rothmund-Thomson syndrome (RTS) is an exceedingly infrequent genetic disorder characterized by a multitude of skin findings collectively known as poikiloderma. In normal cells, the RECQL4 gene is involved in DNA replication and repair. RTS is caused by a mutation in the RECQL4 gene, which results in increased predilection to develop various malignancies. Osteosarcomas and skin cancers are typically associated with this syndrome. We present a rare case of signet-ring cell gastric adenocarcinoma in a patient with RTS. Rothmund-Thomson syndrome (RTS) is a rare autosomal recessive genodermatosis that presents in infancy. Clinical features include short stature, sparse scalp hair, sparse or absent eyelashes, eyebrows, skeletal abnormalities, juvenile cataracts, radial ray defects, premature aging, and a constellation of classical skin findings, known as poikiloderma. RTS is associated with a predisposition to developing certain cancers, particularly osteosarcoma and skin cancer . To our An 18-year-old female patient with a history of RTS and deafness presented to us with abdominal pain, which had been progressively worsening over the preceding one year. It was associated with nausea, vomiting, early satiety, and progressive weight loss due to poor oral intake. Previously, her abdominal pain was thought to be caused by gastroesophageal reflux disease. However, a trial of proton pump inhibitor therapy had not improved her symptoms. On physical examination, she was noted to be cachectic. Findings consistent with the diagnosis of RTS included fish-like facies, congenital deafness, widely distributed poikiloderma (areas of hyper and hypopigmented areas giving the appearance of mottling), and telangiectasias. She had moderate abdominal tenderness.Contrast-enhanced CT scan of the abdomen and pelvis showed gastric and colonic wall thickening, small volume ascites, and borderline upper abdominal and retroperitoneal lymphadenopathy. Esophagogastroduodenoscopy (EGD) with endoscopic ultrasound EUS revealed significantly abnormal mucosa of the gastric cardia. Findings included variegation in mucosal appearance, ulceration, and thickening, highly suggestive of a diffuse infiltrative process with circumferential extension to the level of the pylorus. Figures Gastric biopsies confirmed poorly differentiated adenocarcinoma with signet-ring cell features. Staging laparoscopy showed no peritoneal involvement. She was provided enteral nutrition via jejunostomy tube in preparation for the surgery. Subsequently, she underwent a total gastrectomy with lymph node dissection. R1 resection was achieved with positive proximal and distal surgical margins. Lymphovascular invasion and lymph node involvement  were observed in the surgical specimen. Pathologic stage was finalized as IIIB (pT3N3aM0) with histologic grade G3 (poorly differentiated).Because of poor baseline performance status and severe protein-calorie malnutrition, she was deemed unfit for adjuvant chemotherapy. After a detailed discussion with her family, it was decided to closely monitor the patient with serial body imaging. Unfortunately, her postoperative course was complicated by recurrent episodes of large bowel obstruction, eventually requiring surgical bypass of the obstruction by colo-colostomy (side to side anastomosis of the cecum to the sigmoid colon). Intraoperatively, there was evidence of peritoneal recurrence of gastric cancer, which was confirmed on biopsy of one of the serosal lesions. Her postoperative course was complicated by the development of bilateral pelvic abscesses and septic shock. She underwent CT-guided insertion of the pelvic drainage catheters. Despite our best efforts to treat the infection, her clinical condition continued to worsen. Ultimately, she was transitioned to the inpatient hospice unit.Rothmund-Thomson syndrome (RTS) is a very rare genetic disorder. To date, only 300 cases have been reported in the medical literature . It is tRTS was first described by the German ophthalmologist, Rothmund in 1868 and later in 1936 by an English dermatologist, Thomson . HyperkeAn extensive literature search revealed only one case of gastric carcinoma in a patient with RTS, which was reported in 1975 . One othOwing to the rarity of Rothmund-Thomson syndrome (RTS), routine cancer screening may not be feasible in patients suffering from this congenital disorder. However, our case highlights the importance of recognizing the association of RTS with various malignancies to promptly initiate diagnostic workup based on pertinent clinical presentation. Such an approach may help improve outcomes for this patient population.


Lipomas are benign soft tissue neoplasms of mature adipose tissue commonly occurring in the trunk or extremities. But, intraoral lipomas are rare entities which may be only noticed during routine dental examinations. Especially intramuscular lipomas on the tongue have been reported very rarely. In this study, we report a case of intramuscular lipoma on tongue, with a review of the literature from 1978 to 2019, providing data on age, gender, location, presenting symptoms, size, surgical methods, and recurrence.A case of intramuscular lipoma occurring in tongue region in a 65-year-old male is reported. Surgical excision is the mainstay of treatment for the lesion. In order to decrease the deformity and discomfort after the excision, we tried to modify surgical technique using enveloped mucosal flap. This technique provided more comfortable healing procedure on the operative site without recurrence.This is a rare case of large intramuscular lipoma on tongue. Surgical excision with enveloped mucosal flap design was performed to diminish postoperative raw surface and discomfort and a 24-month follow-up showed excellent healing without any recurrence. A case of intramuscular lipoma on tongue and relevant literature reviews are presented in this study. Lipoma is the most common benign tumor seen as a common entity in the trunk or extremities, accounting for 50% of all soft tissue neoplasm , 2. It oSoft-tissue lipomas can be categorized into two subtypes. Usually, lipomas arise in subcutaneous tissues  . But, thCommonly, surgical excision has been considered for the treatment method of lipoma. However, when the size of the lesion is large, it is likely to cause aesthetic side effects due to the defects after surgical excision. Surgical excision with envelope flap design was performed to diminish postoperative raw surface and discomfort. Here, we report a case of intramuscular lipoma of the tongue which was treated by surgical excision with the envelope technique and the review of literature. A Google Scholar search for the terms “intramuscular lipoma,” “infiltrating lipoma,” “tongue,” and “lingual” within a period from 1978 to 2019 was performed, total 22 literatures published in English –36.A 65-year-old male nonsmoker presented with a painless mass on the right lateral side of his tongue Fig. . This leMagnetic resonance imaging (MRI) showed a 2 cm × 4 cm well-remarked lesion within the muscles of the tongue. The findings were reported as consistent with lipoma .Lipomas are common soft tissue benign tumors, but are rare in the oral cavity, and especially on tongue. The incidence is very low –11. InfrIn order to analyze the epidemiologic and clinical features of intramuscular lipoma developed on tongue, we have searched the literature published in English so far by Google Scholar and reviewed all case reports including our case Table .Since the first intramuscular lipoma on tongue was reported by Bennhoff et al. in 1978, the total of 24 cases have been reported in 23 literatures in English, including present case. Age distribution was shown from 37 to 75 years, and most of the patients over 50 years old (only 3 cases under 50 years). With regard to gender distribution, prevalence of the males was almost twice as high as that of females with 15 cases for male and 9 cases for female. As for the site of occurrence, the lateral side of the tongue was the majority with 16 cases including our case, followed by ventral surface with 4 cases, dorsal surface with 2 cases, and anterior tip with 2 case. The lesion size varied from 0.6 to 10 cm in maximum diameter with an average of 3.18 cm. Because the lesions are typically round or fusiform in shape, the size was analyzed based on the maximum diameter. In most cases, the patient had asymptomatic, painless swelling lesions, but in one case, painful swelling and difficulty in deglutition were present. In three cases, speech difficulties were found. Excision was performed as a treatment method in all cases. Except for six cases without information in the literature, there are no recurrences after surgery in all other cases during the average follow-up period of 17.29 months. Most of the features were not significantly different from simple lipoma of the oral cavity, but in gender distribution, intramuscular lipoma on the tongue was more prevalent in male, whereas simple lipoma was higher in female [Clinically, intramuscular lipomas present as slow-growing, painless, rubbery, and swelling masses. Pain is an uncommon symptom. As the tumor grows in size, functional limitations can arise. Especially on tongue, it can cause discomfort on speech, deglutition, or mastication , 27, 29.The exact etiology of intramuscular lipoma is still unclear. Trauma and chronic irritation have been suggested as probable causes, and also genetic factors and hormonal imbalance are suggested to be related , 38, 39.Imaging such as the computed tomography (CT) scan or MRI plays an important role in making a diagnosis of intramuscular lipoma. In addition, preoperative imaging analysis is essential for defining the size, border, and location of the mass, and relationship with surrounding anatomical structures. Based on them, a surgical plan should be established. Imaging features on CT scan of intramuscular lipoma indicate a lesion situated within the muscle and thick and thin soft tissue density streaks inside a fat density lesion. MRI is also useful method for identifying lipomatous lesions. On MRI, the adipose tissue in the intramuscular lipomas appears as strikingly high intensity signal area on both T1- and T2-weighted images , 40, 41.Intramuscular lipomas have typical histological features. They are histologically well-demarcated but unencapsulated, and have mature uni-vacuolated adipocytes irregularly infiltrating adjacent muscle fibers. No lipoblasts are identified. Also there is no increased mitosis, nuclear atypia, pleomorphysim, cellular hyperchromatism, and lipoblastic proliferation. They show the vasculature composed of thin-walled capillaries, which is occasionally not observed due to compression by surrounding adipocytes , 35, 41.Meanwhile, what should be noted about intramuscular lipomas is that their clinical, histological and imaging characteristics are similar to well-differentiated liposarcomas, malignant tumors. This makes the differential diagnosis difficult , 42. TheThere are differences in imaging features between liposarcomas and intramuscular lipomas. On CT scans, liposarcomas, unlike intramuscular lipomas described above, are characterized by lesions spreading in the intra- and intermuscular layers and fat density lesions with a fair hazy amorphous areas and soft tissue density septa. And they are more oval shaped compared to intramuscular lipomas . On MRI,Due to their infiltrating tendency, the recurrence rate of intramuscular lipomas is known to be higher than that of other subtypes of lipoma. The recurrence rate after treatment ranges from 3 to 62.5% depending on the researchers , 40, 43.When the lesion is large, surgical excision on the tongue lesion can result in the large defect, delayed healing, and patient discomfort. Primary suture was difficult and morphological deformation was predicted due to the large lesion. In this case, we opted for the enveloped flap design because the lesion was 4 cm large. In order to diminish the patient discomfort and reduce healing period, we used surgical excision with enveloped mucosal flap Fig. b. EnveloAlthough very rare, intramuscular lipomas can arise on tongue. Adequate preoperative examinations for accurate differential diagnosis, especially well-differentiated liposarcoma, and appropriate surgery are required to prevent recurrence. In our case, we performed MRI, biopsy before operation. Surgical excision with envelope flap design was performed to diminished postoperative raw surface and discomfort, and a 24-month follow-up showed excellent healing without any deformation and malfunction. Recurrence was not observed for a follow-up period, but long-term follow-up should be required.


Uterine tumors resembling sex-cord tumors are a rare group of tumors with uncertain etiology and histogenesis. The sex-cord tumors are classified into two groups. The first group includes endometrial stromal tumors with foci of sex cord differentiation less than 50% while the second group is composed predominantly or exclusively by sex cord-like elements. We report the case of a middle-aged woman who presented with heavy vaginal bleeding with initial ultrasound findings suggestive of uterine leiomyoma. There was no improvement noticed after a trial of medical treatment; hence, the surgical treatment in the form of total abdominal hysterectomy was undertaken. A few weeks after the surgery, the patient presented with unexplained abdominal pain. Imaging studies demonstrated a hypermetabolic lesion in the upper part of the vagina that was suspicious for malignancy. Complete resection of the mass was performed along with para-aortic lymphadenopathy. Histopathological examination revealed a uterine tumor resembling a sex-cord tumor. Uterine tumors resembling sex-cord tumors are a unique group of uterine neoplasms that exhibits diverge clinical and biological characteristic. Surgical pathologists must recognize this rare entity and differentiate it from other lesions. Uterine tumors resembling sex-cord tumors are a rare group of tumors accounting for less than 0.5% of all uterine malignancies . These tA 41-year-old woman presented to our gynecology clinic with a long history of heavy menstrual bleeding. The bleeding was associated with abdominal discomfort. There was no history of bleeding from any other sites. She did not complain of generalized fatigue, anorexia, or weight loss. Her medical history is remarkable for diabetes mellitus that is well-controlled with anti-diabetic agents. The family history was non-contributory.The patient reported that she had not undergone an investigation for her condition. She was offered medical treatment in the past, but it did not result in any improvement. The results of coagulopathy screening were normal. Transabdominal ultrasonography revealed a hypoechoic uterine lesion suggestive of uterine leiomyoma. In light of the patient’s age and prior unsuccessful medical therapy, she was offered a surgical treatment in the form of a total abdominal hysterectomy. It is worth-noting that the quality of life of the patient was significantly affected as she needed frequent blood transfusions because of her heavy periods.Histopathological examination of the resected specimen revealed a multinodular tumor composed of sheets of cells with uniform oval nuclei and pale cytoplasm Figure . ImmunohA few weeks after the operation, the patient presented with severe lower abdominal pain with no associated symptoms. The pain was not resolved by symptomatic treatment. Abdominal magnetic resonance imaging demonstrated an ill-defined mass, measuring 6.5 cm × 2.6 cm × 2.8 cm, located at the superior part of the vagina with a significant contrast-enhancement. Multiple bilateral pelvic lymphadenopathies were observed. Subsequently, the patient underwent a positron emission tomography scan of an ill-defined hypermetabolic area corresponding to the upper part of the vagina (Figure The findings were discussed in the multidisciplinary oncology meeting and it was suggested that the patient had a local relapse of the tumor and a surgical resection was planned. The patient underwent complete resection of the pelvic mass with para-aortic lymphadenectomy. Histopathological examination of the lesion revealed the diagnosis of uterine tumor resembling ovarian tumors. However, the lymph nodes showed no evidence of malignancy. The patient was discharged and was symptom-free at the follow-up visits 24 months after the initial presentation.We described a rare case of uterine tumor resembling a sex-cord tumor in a middle-aged woman with heavy menstrual bleeding. Generally, this tumor displays a benign biological behavior. It usually affects women in the fourth to sixth decade of life . While iThe exact pathogenesis of this tumor remains unclear. Several postulated theories have been suggested to explain the origin of this tumor. For example, the tumor may be originated from undifferentiated mesenchymal stem cells or from ovarian sex-cord cells that have been displaced during embryological development . As in tConsidering the rarity of the tumor, there are no established treatment guidelines. The management should be discussed in a multidisciplinary meeting by taking the patient’s preferences into consideration . In our Uterine tumors resembling sex-cord tumors are a unique group of uterine neoplasms that exhibits diverge clinical and biological characteristic. Surgical pathologists must recognize this rare entity and differentiate it from other lesions. The tumor should be kept in mind in patients with heavy vaginal bleeding in premenopausal and postmenopausal women. Long-term follow-up is essential because these tumors could have recurrence or relapse.


Textiloma is a retained surgical item such as a sponge or gauze that is unintentionally left in the surgical field after the wound closure. Here, we present the first reported surgical gauze that penetrated the intestine, made a duodenal-ceca fistula and then stuck far away in the ileum. Mechanical obstruction didn't appear clinically or even in radiological investigations because of the fistula which provided the intestinal continuity.We report a 34-year-old man with a previous abdominal interventions complained of cramping, frequent vomiting and presence of undigested food in stool. The frequency of the bowel movement increased recently. Endoscopies, radiological investigations and pathological findings figure out a duodenal-ceca fistula with nonspecific inflammatory tissues in the intestinal biopsy. When we performed the abdomen surgery, retained gauze in the ileum was taken out and the duodenal-ceca fistula was fixed.Gauze or sponge that is forgotten in the surgical field called gossypiboma, textiloma, gauzoma or cottonoid. It could present with various complaints; as an acute or chronic problem, clear or ambiguous symptoms. It could reside in a space; extend across a gap, migrate through a tissue, or even make a fistula between lumina like in our case.Textiloma could change pre-operative diagnosis, intra-operative techniques, postoperative follow-up plan and prognosis. This is the first report proves its ability to mimic inflammatory diseases that penetrate two different lumina and perform fistula. So it should be written in the list of any differential diagnosis when the patient has a previous procedure or surgery. •Textiloma couldn't be always diagnosed preoperatively.•It could give unrelated symptoms to its mass effect or contaminated problems.•It could mimic Crohn's disease clinically, radiologically and even pathologically.•It is a deferential diagnosis for every patient who has a previous procedure or surgery.•Exploring the whole entire abdomen is necessary before performing the techniques. TextiloIn our manuscript, we are going to present textiloma in which the gauze totally migrated into the intestine and left a duodenal-cecum fistula at the point of entrance; this fistula hid the symptoms of mechanical obstruction that happened in the ileum where the gauze was eventually stuck.2A 34-year-old male referred to our university clinic with a complaint of cramping and presence of undigested food in stool. The frequency of the bowel movement increased recently. Frequent vomiting was also developed. The patient denied bloody stool, weight loss, fever or chills.In the past, the patient had open appendectomy five months ago, laparoscopic cholecystectomy seven months ago and open varicocelectomy twenty years ago. Past medical history, family medical history, psychosocial history and drug were unremarkable. His routine labs were unremarkable, and the abdominal ultrasound had no specific findings. Upper gastrointestinal series proved an enterocolic fistula . Abdomin3Textiloma is reported in one out of 1000–15000 intra-abdominal operations . The ris4Gauze that is forgotten in the surgical field could present later with various symptoms in days, months or even years. It is not uncommon to make the diagnosis by an exploratory surgery. Textiloma could change any pre-operative diagnosis, so it should be in the list of any differential diagnosis when the patient has a previous procedure or surgery.Informed consent was taken for this case report. Our study ethical aspects were reviewed and approved by Damascus University deanship, Damascus, Syria.No funding was received for this study.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.All the authors made an equal contribution to the creation of the presented clinical case.Abdul Rahman Hammadieh: the assistant professor and the chief surgeon who prepared the patient and run the operation. ahammadieh@yahoo.com.Mhd Belal Alsabek: attending Surgeon, corresponding author, collected the data, reviewed the PubMed Library and wrote the manuscript. drsabekb@gmail.com.Sara Rustom: undergraduate medical student who participated in writing the manuscript. cu.ty.sara@hotmail.com.1.Name of the registry: This case report is not a first time of reporting new device or surgical technique. So I would not need a Research Registry unique identifying number (UIN).2.Unique Identifying number or registration ID:3.Hyperlink to your specific registration (must be publicly accessible and will be checked):It is a case report not a research.Mhd Belal Alsabek.Not commissioned, externally peer-reviewed.The authors declare that there is no conflict of interests regarding the publication of this paper.


We describe a case of spontaneous partial segmental thrombosis of the corpus cavernosum (PSTCC).PSTCC is a rare condition in which thrombus formation occurs in the corpus cavernosum. This condition is managed in conjunction with a urologist, and management typically includes anticoagulation and pain control. A 39-year-old man presented to the emergency department for two days of worsening pain and swelling to the base of his penis. The patient denied trauma or a history of coagulopathy, had a non-contributory sexual history, and no recent use of erectile dysfunction medications. Examination demonstrated mild swelling to the penile base without evidence of hernia, infection, or shaft injury.Computed tomography (CT) revealed penile asymmetry . UltrasoPSTCC is a rare condition that manifests as penile or perineal pain and swelling. Thrombus formation likely arises secondary to microtrauma, thrombophilia, hemoglobinopathies and, rarely, medication side effect.3What do we already know about this clinical entity?Partial segmental thrombosis of the corpus cavernosum (PSTCC) is a rare condition classically diagnosed with ultrasound or magnetic resonance imaging.What is the major impact of the image(s)?Although computed tomography (CT) has not been previously recommended for identifying this pathology, our case demonstrates that PSTCC can be clearly identified with CT.How might this improve emergency medicine practice?This example of a rare pathology that may go unrecognized by emergency providers demonstrates the use of CT to aid in diagnosis.


Wallerian degeneration (WD) can occur in different projecting systems, such as corticospinal tract, dentate-rubro-olivary pathway, and corticopontocerebellar tract. However, the co-occurrence of hypertrophic olivary degeneration (HOD) and middle cerebellar peduncles (MCPs) degeneration secondary to unilateral pontine infarction in a single patient is extremely rare.A 71-year-old man presented with acute onset of dizzness, slurred speech, and right-sided weakness. On the next day, his previous neurologic deficits deteriorated. Brain magnetic resonance imaging (MRI) revealed acute ischemic stroke of the left pons. After treatment with thrombolysis, antiplatelets, and rehabilitation training, his speaking and motor function improved moderately. At the 3-month follow-up, the MRI showed hyperintensity in the left medulla oblongata and bilateral MCPs on T2-weighted and FLAIR images, suggesting HOD as well as MCPs degeneration.It is of great importance for us to know the anatomic knowledge of dentate-rubro-olivary and corticopontocerebellar pathways. Wallerian degeneration (WD) refers to the progressive anterograde disintegration of axons and accompanying demyelination following injury to the axon or cell body. It can be caused by a wide spectrum of diseases, such as cerebrovascular diseases, neoplasms, hemorrhage, surgery, epilepsy, and white matter diseases . As its A 71-year-old man (weight 60 kg) with known long-standing hypertension and smoking presented with acute onset of dizzness, slurred speech, and right-sided weakness for 1.5 h. Upon admission, the blood pressure was 170/100 mmHg, and the heart rate was 80 beats/min and regular. Neurological examination revealed dysarthria, left facial paralysis, hemiparesis and thermal/pain hypesthesia in his right limbs. The score on the National Institutes of Health Stroke Scale (NIHSS) was 5. An noncontrast computed tomography (CT) of head showed normal findings. Thrombolysis was initiated with 54 mg intravenous tissue plasminogen activator (rt-PA) 2 h after onset. After rt-PA, his previous neurologic deficits deteriorated . Brain MRI disclosed acute ischemic stroke of the left pons  in the medulla oblongata is consistent with hypertrophic olivary degeneration (HOD), which usually occurs following a lesion involving the dentato-rubro-olivary pathway within the triangle of Guillain and Mollaret (GMT) . AnatomiIt is worth noting that hyperintensity in bilateral MCP on T2-weighted imaging can also be noted in this case. The MCPs are mainly composed of pontocerebellar tracts that connect the basal portion of the pons with the cerebellum. By routine MRI techniques, normal MCPs demonstrate homogeneous white matter signal intensity. Previously, classification of diseases that manifest as symmetrical lesions on the MCPs includes neurodegenerative diseases, toxic or metabolic diseases, cerebrovascular diseases and demyelinating diseases . BilaterMRI sequences have been used to depict changes of WD in different stages, especially for corticospinal tract. Histologic and metabolic features on different stages of WD are in accordance with specific findings on MRI . The firIn conclusion, HOD concomitant with bilateral MCPs WD in the setting of unilateral pontine infarction is extremely rare. It is of great importance for us to know the anatomic knowledge of dentate-rubro-olivary and corticopontocerebellar pathways.


Seminoma is the most common subtype of testicular cancer and occurs most commonly in patients aged 30–49 years, but decreases to a very low level in men in their 60s or older.A 90-year-old Syrian man with a 6-year history of an increase in size of his right scrotum, presented to the urological clinic and, on clinical examination, the findings suggested testicular tumor. After orchiectomy and histology results based on microscopic and immunohistochemical examinations, a pure seminoma was diagnosed, so we describe in this case report the second-oldest patient with classical seminoma in the medical literature.This case report has been written to focus on the probability of any type of testicular tumor occurring at any age or decade; urologists should consider seminoma as a differential diagnosis with any testicular swelling even in elderly patients. Testicular cancers account for approximately 1–1.5% of all cancers in men and 5% of urological tumors .Testicular cancers include germ cell tumors (GCTs) 90–95% and non-germ cells tumors (NGCTs)10–15%. GCTs are mainly categorized into unclassified type; seminoma , and non-seminomatous-type tumors include embryonal carcinoma; yolk sac tumor; choriocarcinoma; other trophoblastic tumors; teratoma ; and mixed tumors , 4.Seminoma is the most common testicular germ cell neoplasm and accounts for about 50% of all GCTs, which occur most commonly in patients aged 30–49 years [Less than 20% of patients with stage I seminoma will have retroperitoneal lymph node metastatic disease .In this case report, we describe a classic seminoma that was diagnosed in a 90-year-old patient, who is the second-oldest patient with this condition in the medical literature.A 90-year-old Syrian man with a 6-year history of an increase in size of his right scrotum, without pain presented to the urology clinic with enlargement of, and slight pain in, his right testis. He is a farmer with seven sons and has been smoking for 55 years. In his medical history, he had controlled hypertension and no history of testicular trauma or inflammation. A clinical examination showed the presence a painless, massive enlargement occupying his right scrotum, impermeable to light and with no inguinal lymph node palpable. His vital signs were within normal limits. A scrotal ultrasound scan showed a hypoechoic solid mass measuring approximately 10 cm in his right testis. Laboratory test results revealed tumor markers: alpha fetoprotein (AFP) of 3.1 KU/L , the beta subunit of human chorionic gonadotropin (βhCG) of 2.38 IU/L  and lactate dehydrogenase (LDH) of 420 IU/L .Routine blood test results were within normal limits. The findings suggested testicular tumor and a computed tomography (CT) scan of his abdomen and pelvis was performed and showed no retroperitoneum metastases. His right testicle was removed radically by a right inguinal approach. The pathology test result showed a tumor that is very rare in elderly patients, a classical testicular seminoma. A histological study revealed a nodular solid mass measuring 95*80 mm, with a solid gray cut surface with focal necrosis friable in consistency. The tumor had invaded the hilar soft tissues and tunica albuginea, but without tunica vaginalis invasion. Consequently, the stage was pT2, which it is when the tumor is limited to the testis (including rete testis invasion) with lymphovascular invasion or when the tumor invades the hilar soft tissue or epididymis or penetrates the visceral mesothelial layer covering the external surface of tunica albuginea, with or without lymphovascular invasion. Histological sectioning showed sheets of large polygonal cells Fig. , which sMalignant tumors in the testis are rare. They include GCTs (seminoma and non-seminomatous-type tumors), which account for 90–95% of all primary testicular tumors, and sex cord-stromal tumors  .Classic testicular seminoma and non-seminoma tumors are not common in patients older than 60 years, while spermatocytic seminoma, malignant Leydig cell tumors and lymphomas, in addition to paratesticular sarcomas are more common among elderly patients .The oldest case in the literature is a 92-year-old patient with classic seminoma [Consequently, our patient is considered the second-oldest age in the literature, demonstrating that there is no age limit for this type of malignancy.The patient often presents with a painless, slow-growing enlargement. Acute pain in the testis is noticed in about 10% of all cases and may be due to intratesticular hemorrhage or infraction, and in most cases a testicular mass is noticed. The mass is hard and the skin should be easily separable from it and testicular tumor may be accompanied by or covered with a hydrocele , 10.Transillumination of the scrotum should be performed to distinguish between them and palpation of the abdomen may reveal a bulky retroperitoneal mass. Examination of supraclavicular, inguinal, and scalene nodes should be done also to detect any enlargement .Serum AFP levels are not significantly elevated in patients with seminoma; one study reported very modest elevations in AFP levels in patients with histologically pure seminomas who had a typical clinical course. Serum hCG is often increased in the 10–20% of tumors with admixed syncytiotrophoblast cells. These levels are usually not in excess of 1000 mL U/mL, although one very large seminoma with abundant syncytiotrophoblasts had an associated hCG level of 4000 mL U/mL. Serum LDH is increased in about 80% of patients with advanced-stage disease .Scrotal ultrasonography should be performed rapidly to assess for testicular tumor and to distinguish it from epididymal pathology. This technique may help to check for testicular tumor in the presence of a hydrocele .CT scans of the abdomen and pelvis are used to assess for metastatic disease, especially in the lungs and retroperitoneum .Seminoma is very radiosensitive and most of stage II seminomas are cured with radical orchiectomy and radiotherapy for retroperitoneal lymph nodes, while patients with advanced seminoma should receive primary chemotherapy. Seminomas are sensitive to platinum-based courses and 90% of patients with advanced disease respond to chemotherapy. Most of the residual retroperitoneal masses following chemotherapy are often fibrosis .A positron emission tomography scan should be performed in a patient with a residual mass and, if positive, surgical resection is warranted .In general, testicular tumors should be regularly followed up every 6 months for the first 3 years. During the follow-up, a careful examination of the other testis, the abdomen, and lymph nodes should be done and laboratory investigations, including AFP, hCG, and LDH levels, carried out.Chest X-ray and an abdominopelvic CT scan are used less frequently to detect the risk of relapse in the retroperitoneum .We describe in this case report the second-oldest patient (at 90 years old) diagnosed with classic seminoma in the medical literature. Therefore, classic seminoma could present at any age and should be considered as a differential diagnosis for any painless mass in the scrotum.


Mediastinal hemangiomas are a rare entity frequently developing in the anterior mediastinum. They may exhibit an infiltrating appearance into the surrounding tissue, and extensive surgery is often required despite its benign nature. We report a case of a mediastinal hemangioma mimicking an invasive tumor growth requiring a combined resection of the lung and diaphragm.An asymptomatic 73 year-old-man presented with a 50 mm-sized mass on his chest radiography. A combined resection of the mediastinal mass with the part of the lower lobe of left lung and diaphragm not necessitating a patch repair was performed. The pathological findings were compatible with a diagnosis of a mediastinal hemangioma. Only fibrous adhesions were observed between the tumor and resected lung and diaphragm without any histological invasion.While imaging examinations play a key role in determining a preoperative diagnosis, mediastinal hemangiomas may pose a diagnostic challenge mainly due to its rarity. With a clinical suspicion of a hemangioma during the diagnostic work up, a dynamic CT might be helpful.In mediastinal hemangiomas, a preoperative diagnosis is essential in order to avoid extensive surgery. A hemangioma should be raised as a differential diagnosis for anterior mediastinal tumors, especially in cases with an infiltrative appearance that suggests the necessity for a combined resection of the surrounding organs. They 2An asymptomatic 73 year-old man with a remote history of surgically treated gastric cancer at the age of 40 presented with a 50 mm-sized mass on a chest radiography . He had With the patient in the right semi lateral decubitus position, a total of 3 ports were placed on the 6th, 7th, and 8th left intercostal anterior axillary lines, respectively. Under Carbon dioxide insufflation at a pressure of 8 mmHg, the lesion was visualized adjacent to the pericardium. The lesion was also observed to have grossly invaded into the diaphragm and lower lobe of the left lung. He underwent a total tumorectomy with a combined resection of the pericardial fat, left lung, and diaphragm. Since a complete resection was achieved, no intraoperative frozen section examination was performed. The postoperative course was uneventful and the patient was discharged on the third postoperative day. The macroscopic findings of the specimen revealed a circumscribed mass embedded in the fat tissue . The his3Mediastinal hemangiomas are rare neoplasms accounting for 0.5% of mediastinal tumors and are frequently found in the anterior mediastinum ,2．They hWhile most cases are generally visualized with well circumscribed masses ,8,9, somOn MRI, a fat component and high intensity on the T2-weighted images have been reported as characteristic of a mediastinal hemangioma and likewise of hepatic hemangiomas ,13. PartCalcified phleboliths derived from organized thrombi are the most diagnostic sign, and are detected more sensitively by CT than conventional radiography ,17. HoweOn the other hand, it has been reported that peripheral puddle images showing a punctate contrast effect on the margin of the tumor in the aortic phase on the dynamic CT is highly specific for hepatic hemangiomas , and its4Mediastinal hemangiomas are a rare entity frequently developing in the anterior mediastinum. They could exhibit an infiltrating appearance and often are amenable to extensive surgery despite being totally benign in nature. Therefore, a preoperative diagnosis is essential to avoid extensive surgery. A hemangioma should be raised as a differential diagnosis of anterior mediastinal tumors, especially in cases with an infiltrative appearance suggesting the necessity for a combined resection of the surrounding organs.The authors report no declarations of interest.Not applicable.Not applicable.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.Shogo Yobita wrote this paper. All authors read and approved the final manuscript.Not applicable.Toru Nakamura.Not commissioned, externally peer-reviewed.Not applicable.


Castleman disease (CD) is a rare lymphoproliferative disease with a poorly understood etiology. The occurrence of CD in the abdominal cavity is very rare, especially in the retroperitoneal peripancreatic region.A 33-year-old woman was referred to our department on March 1, 2018 for a detailed physical examination due to retroperitoneal peripancreatic lymph node enlargement over 15 days.Enhanced magnetic resonance imaging of the epigastrium showed the mass with abundant blood supply is located between the liver and the stomach in the upper margin of the pancreas. Postoperative pathological examination revealed CD, type of unicentric Castleman disease.We performed an open surgery on this patient and completely removed the mass. There was no postoperative radiochemotherapy.The patient was followed-up for more than 12 months after the operation and showed good recovery.CD is a rare disorder that is hard to diagnose early and complete resection of the tumor is still the most effective treatment. In addition, it can be classified into 3 histopathological patterns: hyaline-vascular (HV) type, plasma cell (PC) type, and mixed variant. Type of HV is the most common. CD, which develops in all lymph nodes, especially in the mediastinum, can also occur in the cervical, retroperitoneal, and axillary regions. However, invasion into the retroperitoneal peripancreatic region is quite rare.,8 We herein report a rare unicentric Castleman disease (UCD) located in the superior margin of the pancreas.Castleman disease (CD) is a rare nonclonal lymph proliferative disorder of unknown etiology, which was first described as a pathologic entity in 1954 and later defined by Dr Benjamin Castleman in 1956.2A 33-year-old woman was referred to our department on March 1, 2018 for a detailed physical examination due to retroperitoneal peripancreatic lymph node enlargement over 15 days and she had no apparent discomfort. The patient had no significant medical history and had no significant family history. The physical examination showed no positive signs.Laboratory examinations revealed that the patient was negative for the human immunodeficiency virus (HIV) antibody. Karyotype analysis of bone marrow chromosomes revealed 10 normal metaphases and the results of bone marrow immunotyping (CD series) – lymphoma (type to be determined) showed no abnormal phenotypes in NK and T lymphocytes, and no clonal abnormalities in PCs and B lymphocytes. Classification of bone marrow cells, tumor markers, and other laboratory test results were also within the normal range. Enhanced magnetic resonance imaging of the epigastrium showed the mass with abundant blood supply is located between the left lobe of liver and the stomach in the upper margin of the pancreas , CD23(+); interfollicular zone cells CD3(+), CD43(+), ki67(+)10%; follicular germinal center cells CD20(+), CD79ɑ(+), ki67(+)70%, blc-2(−), CD10(+), bcl-6(+)  reexamination on March 4, 2019  cases. However, CT, MRI, and ultrasound can still provide reference values for the diagnosis of retroperitoneal UCD. On CT imaging, retroperitoneal UCD is generally a well-defined mass with different morphologies, such as elliptical, kidney-shaped, and dumbbell-shaped, which can easily be distinguished from a spherical paraganglioma. The plain CT scan images of this disease show low or equal density lesions. The majority of lesions exhibit inhomogeneous enhancement on enhanced CT images. In addition, some tumors have a rich vascular supply, accompanied by Microcalcifications. On the magnetic resonance imaging (MRI), most retroperitoneal UCD lesions show an isointense or hypo-intense signal on T1WI, and a slight hyperintense or hyperintense signal on T2WI. In addition, ultrasound also has some advantages in estimating the location of the mass and its adjacent feeding vessels and a homogeneous hypoechogenic formation is commonly shown on ultrasound.Because the tumors are located behind the peritoneum, we cannot perform preoperative biopsy. So, retroperitoneal peripancreatic UCD is usually concealed and difficult to diagnose. The radiological appearance of CD is nonspecific. Imaging manifestations of the disease are very difficult to distinguish from other diseases, including neuroendocrine tumor, paraganglioma, or lymphoma. The percentage of HV type and PC type is approximately 90% and 10%, respectively. Mixed type cases are rarely reported. Most HV type cases manifest as UCD, while PC type cases usually occur in the form of multicentric CD. The characteristics of the hyaline vascular type include considerable enlarged lymphoid follicular proliferation at various levels of maturity. In addition, lymphoid follicles are normally scattered throughout the tissue. The PC type is almost related to the multicentric form of CD. This type shows less vascularity and is characterized by sheets of mature PCs within the interfollicular tissues surrounding larger germinal centers. Histological examination of our case revealed enlarged lymph nodes consisting of multiple follicles of different sizes, degeneration of germinal centers and hyaline degeneration of blood vessels. High power microscopy showed that the follicles were surrounded by concentric rings of lymphocytes.Histological evaluation plays a vital role in the exact diagnosis of CD. The histopathological features consistent with CD diagnosis include abnormal regressed or hyperplastic germinal centers, follicular dendritic cells prominence, hypervascularisation, expanded mantle zones, and interfollicular plasmacytosis. Two major pathological features can be defined: a HV subtype and a plasmacytic (PC) subtype, with some lesions exhibiting mixed characteristics. Complete resection provides confirmation of the diagnosis with histological evaluation. If complete surgery resection is not possible, debulking of the main lesion can be considered to reduce local symptoms. Residual associated small lymph nodes may involute after surgery. Radiotherapy and chemotherapy can be used for unresectable or recurrent tumors. But their efficacy still needs to be confirmed by further studies. At present, there is no standard protocol for predicting the prognosis and effectively managing UCD. In asymptomatic patients for whom surgery is impossible or too mutilating, watch-and-wait can be considered. Our patient underwent complete resection of the tumor and did not undergo radiochemotherapy after surgery. In this report, we described 1 rare case of retroperitoneal peripancreatic UCD, which will contribute to an improvement in the understanding of UCD and provide a significant reference for future cases.Surgery remains the gold standard if the lesion is amenable to complete resection.In this study, no special individuals and organizations need to be appreciated.Resources: Sheng Wu.Supervision: Yong Ji, You-Xin Zhou.Writing – original draft: You-Xin Zhou.Writing – review and editing: You-Xin Zhou.You-Xin Zhou orcid: 0000-0001-8498-709X.


Pleomorphic adenoma is a common benign salivary gland tumor which presents as a painless swelling that gradually increases in size, if left untreated. It is often seen involving the parotid gland. However, pleomorphic adenoma has been reported to involve the minor salivary glands as well. In this report, we present two cases of pleomorphic adenoma originating from minor salivary glands of the upper lip, occurring in two male patients of the same age (44 years) with markedly similar history of duration, size, and site of the lesion. The tumor was excised in both patients and sent for histopathological analysis which showed features of pleomorphic adenoma confirming the diagnosis. Pleomorphic adenoma is a common benign tumor of the salivary gland. These lesions account for two-thirds, or approximately 60%-65%, of all salivary gland tumors . It showIn this paper, we present two cases of pleomorphic adenomas involving the minor salivary glands, with strikingly similar clinical and demographic characteristics.Case 1A 44-year-old male patient visited the outpatient department (OPD) with a painless solitary growth intraorally in the upper right lip since five years. The growth initially appeared as a small lesion, which eventually grew to its present size. On examination, we found a well-defined, round, firm, non-tender, non-fluctuant, and mobile lesion with a size of approximately 3 × 3 cm on the right side of the upper lip. Class II gingival recession in relation to the right canine was noted. The overlying mucosa was pinkish in color, with evidence of superficial vascularity. Figure Case 2A 44-year-old male patient reported to the OPD with a solitary swelling on the left side of the upper lip since five years, with intermittent pain in the region. The lesion started as a small growth, which subsequently increased to its current size. On examination, the mass was found to be a well-defined, round, firm, non-tender, non-fluctuant and mobile lesion of size approximately 4 × 3 cm on the left side of the upper lip. The overlying mucosa appeared normal, with superficial vascularity. Preoperative view of the tumor for Case 2 can be seen in Figure Surgical excisionThe same surgical procedure was used in both the cases. A vestibular incision was made over the lesion as seen in Figure Pleomorphic adenoma is a tumor that is often associated with major salivary gland especially the parotid gland. However, its involvement with minor salivary glands has been described as well. Shreshta et al. stated in their study that pleomorphic adenoma with respect to the upper lip is more common than those of the lower lip by a ratio of 6:1 . Kroll aPleomorphic adenoma is essentially a benign tumor. Nevertheless, aggressive behavior of the lesion has been described; there have been instances of the tumor invading neighboring vessels in the absence of any other features of malignancy . In theiPleomorphic adenoma is a benign neoplasm most commonly seen originating from the parotid gland. Here, we reported two unusual situations of pleomorphic adenoma involving the minor salivary glands with identical clinical and demographic characteristics with respect to location, size, and history of the lesion. Such an occurrence at the same period of time is quite rare and noteworthy.