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We present a case of catatonia in a female in her 70s that was successfully managed with zolpidem. This report highlights the challenges in managing treatment-resistant depression with catatonic features and the importance of a multidisciplinary approach in optimizing patient outcomes.
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Psychiatry
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Our patient was an elderly woman who had first come to the attention of mental health services about four years ago due to an episode of acute-onset depressive features, possibly triggered by work-related stress. Before this, she had no prior history of mental health disorders. Despite trying numerous antidepressants (sertraline, mirtazapine, venlafaxine, vortioxetine, trazodone, and escitalopram), antipsychotics (quetiapine and olanzapine), anxiolytics, and lithium, her depressive symptoms had remained persistent. She had been hospitalised two years ago for a depressive episode with psychotic features characterised by low mood, loss of interest in enjoyable activities, and a preoccupation with food that had led her to stop eating at home. She had also exhibited possible catatonic features, including mutism and unusual behaviours such as repeatedly running up and down the stairs or moving from sitting to kneeling positions. At that time, she had undergone 15 ECTs twice a week with no significant improvement. Vascular depression had been suspected due to treatment resistance and atypical illness trajectory. The Edinburgh Cognitive and Behavioural ALS Screen (ECAS) had also shown dysexecutive features in her cognition. Notably, neuroimaging studies had shown non-specific frontal atrophy on MRI and minor frontal perfusion reduction on SPECT scans. Minimal changes had been observed between the scans conducted two years apart.
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Psychiatry
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She was subsequently admitted to a psychiatric ward for older people for a period of assessment. On mental state examination, she was well-groomed with intense eye contact and exhibited signs of anxiety and depression. She was guarded and showed suspicious behaviour towards the staff. She had good insight into her illness and there were no thought/ perception abnormalities. Physical examination revealed no positive findings. However, her blood tests indicated elevated white cell count with neutrophilia and there were three positive urine cultures for E. coli during the admission. Hence, she was commenced on prophylactic trimethoprim after review with urology. Further assessments by neurology revealed no focal neurological features but they recommended additional investigations including HIV, Lyme, syphilis virology, autoimmune encephalitis screen, and micronutrient levels, which were all unremarkable. Table 1 shows the investigation results.
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Psychiatry
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During the early week of the admission, her distress and agitation escalated, prompting an attempt to leave the ward. She was soon deemed to lack the capacity to give consent to treatment and was detained under the Mental Health Act. She was mostly in her room offering no interaction, with minimal oral intake, though there were short-lived episodes where she presented as bright and would converse freely with staff. During episodes of psychomotor agitation, she was observed to be grinding her teeth (grimacing) and walking in a jerky staggered fashion. Occasionally she was heard wailing, visibly distressed, and would place herself on the floor. Sudden and dramatic clinical responses within minutes were noticed with zolpidem administration for managing these episodes of acute distress. The Antecedent-Behaviour-Consequence (ABC) chart was employed in other to understand the trends and patterns of these episodes. The chart showed that the symptoms were most prominent in the morning and were exacerbated by staff attempts to provide verbal reassurance to her.
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Psychiatry
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During a well-documented episode, she started with loud vocalisations and agitated pacing around the room, accompanied by abnormal posturing and grimacing. Her behaviour included intermittently leaving her bed to pace in a circular fashion with a distinctive gait, alternating between walking on tip-toes and undulating her head and torso. Throughout the episode, she made continuously loud and repetitive moans without coherent speech. Her only verbal communication consisted of echolalia, repeating sentences from nursing staff attempting to reassure her. She also displayed stereotypic movements with the occasional lifting of her hips on the bed.
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Neurology
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Further examination demonstrated that whilst her upper limb tone was normal, she resisted extension of her lower limbs and this was proportionate to the force applied by the examiner (gegenhalten). During the examination, she rose again, displaying wailing and pacing behaviours in the same manneristic fashion. Her BFCRS score was 17 points. It was positive for excitement, mutism, staring, posturing/cataplexy, grimacing, echolalia, stereotypy, mannerism, negativism, withdrawal, impulsivity, and gegenhalten. Following the administration of 10 mg of zolpidem, her BFCRS score decreased to 6 points after 30 minutes, scoring only for mutism, staring, negativism, and withdrawal. She appeared more relaxed following this medication and was able to participate in conversation. At this stage, she struggled to describe her experience in detail, though she had described a build-up of inner tension and some generalised abdominal discomfort, leading to a feeling of “release” during the episode.
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Neurology
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She was started on regular zolpidem as she and her family opted against ECT due to past non-response and hesitancy regarding lorazepam re-prescription. Her dose was gradually increased to 10 mg four times daily. Subsequently, significant improvement was observed as she engaged in outings with her family, ward-based activities, and interaction with other patients. An improvement was noted in her ability to attend to her personal care and her oral intake increased. Venlafaxine was commenced to manage the depressive episode and was gradually titrated to 75 mg in the morning and 37.5 mg at night. Her progress remained stable, leading to her discharge after 10 weeks of inpatient admission. The patient has shown continued improvement and has not required readmission over the past six months. She remains on the same dosage of zolpidem, as previous attempts to reduce the dose had led to concerns about the recurrence of catatonic features.
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Psychiatry
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Few reports on revascularization therapy for this disease are available. Moreover, stent migration and occlusion caused by progression of coronary aneurysm have been reported 9 months after sirolimus-eluting stent implantation for a patient with CAD in IgG4-RD. 3 Therefore, new treatment modalities for IgG4-RD-CAD are needed. Drug-coated balloon (DCB) is one of the leading-edge devices that transfer antiproliferative drugs into the lesions via single balloon inflation to prevent restenosis, leaving nothing implanted in the coronary artery tree. In this report, we describe a case with CAD in IgG4-RD that responded favourably to local treatment with DCB and immunosuppressive agents.
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Cardiology
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The patient was a current smoker (20 cigarettes/day for 50 years) with a history of hypertension and dyslipidaemia. Low-density lipoprotein-cholesterol was well controlled at 64 mg/dL (normal: 65–163) with atorvastatin 10 mg/day. No remarkable findings were noted upon physical examination, and transthoracic echocardiography revealed normal left ventricle function in spite of abnormal Q waves in the II, III, and aVF leads of the electrocardiogram. Coronary computed tomography angiography (CCTA) revealed right coronary artery hypoplasia, coronary artery aneurysm with severely calcified stenosis in the proximal left anterior descending artery (LAD) and subtotal obstruction in the mid-LAD. The left circumflex artery (LCX) showed significant stenosis in the ostium, aneurysmally dilated lesions in the mid and total occlusion in the distal segment. Moreover, diffuse perivascular soft tissue thickening was detected in the mid-LCX. Additional coronary angiography (CAG) also revealed a severe three-vessel disease with aneurysmally dilated lesions both in the LAD and LCX .
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Cardiology
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As both the distal runoff of the LAD and LCX were too poor for grafting under bypass surgery, percutaneous coronary intervention (PCI) was performed for the culprit proximal- and mid-LAD lesions with aspirin 100 mg/day and clopidogrel 75 mg/day. DCB-only angioplasty was considered for these lesions as the patient was suspected of having vasculitis owing to the specific aneurysmal appearance of the coronary artery and presence of diffuse perivascular soft tissue thickening in the LCX. PCI was performed via the right femoral artery. The mid-subtotal lesion in the LAD could not be fully dilated with a 1.5 mm balloon and an intravascular ultrasound (IVUS) imaging demonstrated diffuse fibrosis and fibro-calcified lesions in the distal LAD. Thus, rotational atherectomy for both the proximal and mid-lesion was performed with a 1.5 mm burr (175 000 rpm) (Boston Scientific, USA), followed by debulking of the proximal lesion with a 2.0 mm burr (175 000 rpm). Additional scoring balloon dilatation [Lacrosse NSE® 2.5 mm at 4atm (Nipro, Japan)] for the mid-LAD and DCB angioplasty [SeQuent Please® 2.5 mm at 2atm for the mid-lesion and SeQuent Please® 3.5 mm at 2atm for the proximal lesion (B. Braun, Germany)] was performed, and a smooth lumen without coronary dissections was obtained (see Supplementary material online , Videos S1 and S2 ). The patient’s symptoms resolved, and he was discharged.
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Cardiology
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Blood test results showed positive antinuclear antibodies 320× (normal < 40×), anti-ds-DNA antibodies 196 IU/mL (normal < 12), and anti-SS-A/anti-SS-B antibodies, with positive lymphocytic infiltration in the labial salivary gland histopathology leading to the diagnosis of Sjögren’s syndrome (ACR/EULAR classification criteria). 4 The patient also exhibited elevated IgG and IgG4 (230 mg/dL; normal: 11–121) levels. Because Sjögren’s syndrome is not presumed to be complicated by vasculitis, the rheumatologist diagnosed this case as an overlap between Sjögren’s syndrome and IgG4-RD. Three months after revascularization, the patient developed interstitial pneumonia and was started on oral steroids and immunosuppressive drugs. He is currently undergoing treatment with prednisolone 3 mg/day, tacrolimus 3 mg/day, nintedanib 150 mg/day, abatacept 125 mg/day, and clopidogrel 75 mg/day; his IgG4 levels are normal.
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Rheumatology
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A 42-year-old female presented with features of obstructive jaundice. There was no history of abdominal pain, fever, loss of appetite, or weight loss. On examination, there was icterus and hepatomegaly with no other signs of chronic liver disease. A liver function test (Table 1 ) demonstrated total bilirubin of 14.5 mg/dl (normal range 0.3-1.3 mg/dl) and direct bilirubin of 11.5 mg/dl, (normal range 0.1-0.4mg/dl), Aspartate transaminase (AST) of 103 IU/L (normal range 12-38 IU/L), alanine transaminase (ALT) of 58 IU/L (normal range 4-41 IU/L), and alkaline phosphatase (ALP) of 566 IU/L (normal range 35-130 IU/L). She tested positive for Hepatitis B infection with HBV DNA levels of 602 copies/ml.
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Hepatology
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She subsequently underwent endoscopic retrograde cholangiopancreatography (ERCP) with CBD stenting for rising bilirubin, and the decision was taken to opt for surgical resection. Intraoperatively, there was a cystic lesion of size around 10 x 9 cm at the hilum of the liver, but fortunately, it engulfed only the left hepatic duct, left hepatic artery, and the left portal vein. The lesion involved segments 2,3 and 4, extending onto segments 5,8 of the liver . Mucinous material was noted in the distal CBD, causing proximal dilatation of the bile duct. As the liver was nodular and fibrotic, to preserve the liver parenchyma, a left hepatectomy along with total excision of the cyst was done. The extrahepatic bile duct was excised, and the right cholangiojejunosotomy was done. Her postoperative histopathology revealed intraductal papillary neoplasm with low-grade dysplasia . She is on regular follow-up, and one year after surgery, the patient is symptom-free with no evidence of recurrence.
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Hepatology
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An initial staging laparoscopy ruled out evidence of metastasis, and intraoperatively, a mass of size 10 x 8 cm involving the segments 6, 7, and 8 with an abutment to the inferior vena cava was noted. A decision was taken to proceed with the right hepatectomy, and during parenchymal transection, the lesion was found to have an extension into segment 4 . Right hepatectomy along with cyst excision was done. Postoperative histopathology was consistent with intraductal papillary neoplasm of the bile duct with invasive carcinoma (pT1bN0M0); the resected margin showed features of tumor infiltration. A tumor thrombus was also noted in the bile duct. Immunohistochemistry showed strong membrane positivity for cytochrome 7 (CK7) (>90% tumor cells), cytochrome 20 (CK20)- focal membrane positivity in >20% tumor cells, cytochrome 19 (CK19)-focal positivity in 50-60% cells, and caudal type homeobox 2 (CDX2) was negative . Postoperative events were uneventful, and she was planning on adjuvant chemotherapy (Capecitabine) after the multidisciplinary team (MDT) discussion.
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Hepatology and Surgical Oncology
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A 78-year-old male presented with features of obstructive jaundice. A liver function test (Table 2 ) demonstrated total bilirubin of 19.6 mg/dl (normal range 0.3-1.3 mg/dl) and direct bilirubin of 13.2 mg/dl (normal range 0.1-0.4mg/dl), AST of 79 IU/L (normal range 12-38 IU/L), ALT of 40 IU/L (normal range 4-41 IU/L), and ALP of 349 IU/L (normal range 35-130 IU/L). Upper gastrointestinal endoscopy demonstrated a nodular lesion over the ampulla. The biopsy was negative for malignancy.
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Gastroenterology
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CECT and MRI of the abdomen showed two soft tissue lesions (15.6 x 12 mm and 13 x 12 mm, respectively) in distal CBD. The patient underwent pancreaticoduodenectomy, and postoperative histology showed features suggestive of Intraductal papillary neoplasm of the bile duct with invasive carcinoma . The patient underwent adjuvant chemotherapy after the MDT discussion.
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Gastroenterology and Oncology
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In the above first reported case, there was a diagnostic dilemma as obstructive jaundice was caused by the mucin produced from the IPNB, a lesion that itself is difficult to diagnose preoperatively. With a hepatitis B positive, a marginal liver status, and a tumor at the hilum of the liver, the decision was taken to remove the cyst with parenchymal preservation rather than a formal anatomical resection, as resection was the only chance for cure, apart from a liver transplant that is limited by scarce donor availability. However, the patient must be on follow-up for life as the reported recurrence is high and unpredictable for this rare neoplasm. The second case was IPNB with invasive carcinoma; technical difficulties in operative procedures precluded R0 resection, and the prognosis was poorer compared to the first case. However, she is planned for adjuvant chemotherapy and put on surveillance to decide upon the further plan of management. The third patient had a presentation like a periampullary carcinoma; this shows that the intraductal papillary neoplasm of the bile duct has a wide range of presentations, contributing to diagnostic and therapeutic difficulties.
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Hepatology
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We present the case of a 79-year-old male patient with advanced CA and severe mitral and tricuspid regurgitation receiving TEER of both the mitral valve (MV) and tricuspid valve (TV) as the first reported case in a single patient. This case highlights feasibility and delineates challenges of dual TEER in CA.
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Cardiology
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The 79-year-old male patient was diagnosed with ATTR-CA in December 2022. He experienced persisting symptoms with dyspnoea New York Heart Association (NYHA) class IV despite optimized heart failure (HF) therapy, consisting of beta-blockers, MR antagonists, SGLT2 inhibitors, loop diuretics, and transthyretin-stabiliser therapy with tafamidis. The patient has a history of relevant comorbidities, including coronary artery disease and atrial fibrillation. Echocardiography revealed a concentrically hypertrophied ventricle and enlarged atria. The right ventricular function was mildly impaired, and right heart catheterization showed postcapillary pulmonary hypertension (mean pulmonary artery pressure 31 mmHg; pulmonary capillary wedge pressure 24 mmHg).
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Cardiology
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The patient underwent further evaluation at the West German Heart and Vascular Centre, University Hospital Essen, Germany, which revealed severe MV and TV regurgitation on echocardiographic examination. Six-minute walk test was not possible due to resting dyspnoea, NT-proBNP was elevated with 3826 pg/mL (normal value < 125 pg/mL), and renal retention parameters were within normal range . Due to the patient’s hypotension and limited pharmacological options for HF management, a decision was made to proceed to interventional treatment. Progression of coronary disease was ruled out by coronary angiography. Transoesophageal echocardiogram (TOE) confirmed secondary mitral regurgitation (MR) with a vena contracta (VC) of 10 mm, an effective regurgitation orifice area (EROA) of 0.43 cm², and regurgitation volume of 64 mL . Tricuspid regurgitation (TR) showed a VC of 9 mm, EROA was 0.69 cm 2 , and regurgitation volume was 67 mL . Due to high surgical risk, the patient was planned for a staged interventional approach as per interdisciplinary heart team consensus. Low stroke volume (45 mL) and thickened valve leaflets were identified as intervention-specific risk factors that could complicate procedural success of TEER. The PASCAL-Ace (Edwards Lifescience, California, USA) devices were used for both procedures, which were performed with right femoral vein access with local anaesthesia and moderate sedation ( Supplementary material online , Video S1 ).
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Cardiology
|
The first intervention targeted the MV and was conducted in May 2023 with successful placement of two PASCAL-Ace devices . A satisfactory result was achieved, with only first-degree mitral regurgitation and a post-interventional mean pressure gradient (MPG) of 3 mmHg . No complications such as postoperative bleeding or rhythm disturbances were seen. No relevant shunt fraction from iatrogenic atrial septal defect after transseptal puncture was documented. Oral anticoagulation was continued the day following implantation. The patient was discharged on Day 6 following intervention. High-grade TR did not show relevant changes in follow-up.
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Cardiology
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In August 2023, the patient underwent the second interventional procedure to address high-grade TR with successful placement of two PASCAL-Ace devices . Post-interventional echocardiographic examination showed a minimal residual TV insufficiency with a post-interventional MPG of 3 mmHg . A duplex sonography of the puncture site showed an arteriovenous fistula with a flow volume of 200 mL/min which was considered non-haemodynamically relevant. No further therapy was necessary, and a follow-up 3 weeks later showed stable findings without an increase in flow volume. The patient was discharged 4 days post-tricuspid valve intervention.
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Cardiology
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The patient was closely monitored following both interventions, and a routine follow-up visit was scheduled three months after the last intervention. Here, the patient reported mild residual dyspnoea (NYHA II). Transthoracic echocardiography revealed an MV MPG of 5 mmHg and a TV MPG of 2 mmHg. LV function was quantified at 41%, with all devices in loco typico . Laboratory chemistry revealed a reduction in NT-pro BNP levels at 1649 pg/mL. The 6 min walk test showed a distance of 290 m. The post-interventional haematoma regressed, and the patient continued to report no pain or other related symptoms. The patient is currently under routine follow-ups.
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Cardiology
|
The first patient with a life-threatening event became unresponsive, “was not breathing” (coded with MedDRA PTs unresponsive to stimuli, respiratory arrest), and was given rescue breathing, then responded and became alert. The patient was discharged after an observation period of 150 minutes. The patient’s history of polysubstance abuse and kidney transplant for end-stage renal disease may have contributed to the events. The second patient with a life-threatening event became unresponsive, looked like they were not breathing, and had no pulse (coded with MedDRA PT cardiorespiratory arrest). Cardiopulmonary resuscitation was performed, and the patient’s pulse and respirations restarted. The patient was taken to the emergency department and released on the same day. The patient had a history of syncope, very low blood pressure, hypothyroidism, and of note, used a cardiac loop recorder, indicating a possible cardiac pathology under consideration. Concomitant medication use of lithium, clonazepam, eszopiclone, trazodone, and bupropion might have contributed to the events.
|
Pharmacology/Toxicology
|
On the follow-up contrast-enhanced MRI of the neck, the mass ( arrow ) measures 2.3 × 3.6 × 2.1 cm on MRI of the neck and is homogeneous and isointense to muscle ( arrowhead ) on axial T1-weighted image , and hyperintense to muscle ( arrowhead ) on axial T2-weighted image . These findings were reported as nonspecific, with differentials including conglomerate lymphadenopathy related to a lymphoproliferative process or a mesenchymal tumor such as a sarcoma. Although uncommon, the differential could have included normal thymic tissue in an ectopic cervical location along the expected developmental path from the third and fourth branchial pouch. 1 4 5 The fine needle aspirate with flow cytometry was performed showing mature T-cells, but neoplasm (versus benign thymic tissue) was not excluded. Due to the inconclusive MRI report and the biopsy unable to exclude malignancy, a contrast-enhanced fluorodeoxyglucose positron emission tomography/computed tomography (F- 18 FDG PET/CT) was ordered for further evaluation.
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Radiology
|
A case of extreme RSA was observed in a young, untrained woman. The heart period oscillated between approximately 600 ms during inspiration and 1,100 ms during expiration. The prolonged heart periods during expiration led to an instantaneous BP drop of 25 mmHg. HR and respiration entered a loose coupling, with inspiration being triggered by the heartbeat and expiration activating the previously suppressed vagal cardio deceleration. RSA was primarily centrally mediated, likely due to the inhibition of the baroreflex by inspiratory activity. Maximal RSA excursions were observed at a breathing frequency of 0.167 Hz (10/min), which aligned with her slender build. No cause for the extreme heart rate excursions was identified. No medical follow-up was deemed necessary.
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Cardiology
|
A 50-year-old woman presented with a three-month history of low back pain. Past medical history was significant for hypertension, pulmonary embolism, and cholecystectomy. There was no previous exposure to chemicals. The physical examination was unremarkable. Laboratory studies revealed a white blood cell count of 7.8 × 10 9 /L with 55% neutrophils and 2% immature granulocytes including metamyelocytes, myelocytes, and promyelocytes. She had mild basophilia (4%) and anemia with a hemoglobin of 10.3 g/dL. The platelet count was 269 × 10 9 /L. Her peripheral smear showed rouleaux formation. A complete chemistry panel was unremarkable with normal serum calcium and creatinine. Serum immunoglobulin G (IgG) level was elevated at 4,930 mg/dL. Serum protein electrophoresis revealed an M-protein of 4.2 g/dL, which was IgG kappa (IgG-K) on serum immunofixation. Urine protein and immunofixation electrophoresis showed kappa Bence Jones proteinuria of 606 mg in 24-hour urine volume. Beta-2 microglobulin level was 4.2 mg/L (0.7-1.8 mg/L). The bone survey showed myelomatous lesions in the skull, left scapula, L5 vertebra, and long bones. A magnetic resonance imaging (MRI) of the lumbosacral spine showed an epidural tumor involving the L5 vertebra and myelomatous lesions in the majority of the thoracic and lumbar vertebra. The patient underwent a resection of the epidural tumor. The pathology showed kappa light chain-restricted monoclonal plasma cells. The patient then underwent a bone marrow aspirate and biopsy, which showed megakaryocytic hyperplasia and the infiltration of kappa light chain-restricted plasma cells comprising 70% of the total cellularity , confirming the diagnosis of multiple myeloma (MM). Interestingly, the cytogenetic studies showed two different clones: one hyperdiploid clone with complex karyotype and structural abnormalities involving chromosome 1 in 12/20 metaphases and a clone with Philadelphia chromosome, t(9;22)(q34;q11.2), in 5/20 metaphases . Fluorescence in situ hybridization (FISH) was negative for immunoglobulin heavy chain (IgH) gene rearrangement, monosomy 13 or loss of RB1 locus, and TP53 gene deletion but was positive for a clone with BCR - ABL1 rearrangement as shown in Figure 5 . The quantitative real-time polymerase chain reaction (PCR) analysis of peripheral blood samples detected the presence of BCR - ABL fusion transcripts, thus establishing the diagnosis of chronic-phase chronic myeloid leukemia (CML).
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Hematology
|
The patient received induction therapy for MM with the combination of bortezomib, cyclophosphamide, and dexamethasone and achieved a very good partial response. For CML, the patient received imatinib 400 mg daily and achieved complete cytogenetic and major molecular remission with a reduction in BCR - ABL / ABL ratio from 15.05 to 0.07 after three months. She then underwent peripheral blood stem cell collection after mobilization with granulocyte colony-stimulating factor (G-CSF), followed by high-dose melphalan (200 mg/m 2 ) and autologous hematopoietic stem cell transplantation for MM. The posttransplant course was largely uneventful. An evaluation for MM at day 100 after ASCT showed complete remission. At the last follow-up (two years since diagnosis), the patient remains on imatinib and is in complete cytogenetic and major molecular remission. She opted not to take maintenance therapy for MM but remains in complete remission.
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Hematology
|
In this article, we present a rare case of postoperative vision loss in a 70-year-old patient following a TLIF procedure for degenerative lumbar spine disease. The patient developed complete vision loss in the left eye due to decompensation of pre-existing glaucoma, which was attributed to improper head positioning during the surgery. This case underscores the importance of a thorough preoperative assessment of ocular health in patients with known risk factors, such as glaucoma, and highlights the critical role of intraoperative positioning in preventing ocular complications.
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Neurosurgery
|
Our case report highlights the importance of careful intraoperative management for patients with glaucoma undergoing spinal surgery. The 70-year-old patient had a known history of glaucoma in the left eye, which had been medically managed with eye drops prior to the surgery. However, due to improper head positioning during the TLIF procedure, the patient experienced external compression of both eyes, leading to a rapid decompensation of glaucoma in the left eye. Postoperatively, the patient suffered from complete vision loss in the affected eye, and despite subsequent glaucoma surgery, the vision loss persisted.
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Ophthalmology
|
We presented a 70-year-old male with complaints of left-sided cruralgia, lumbar radicular pain (sciatica-type L5), and right-sided weakness. He reported paresthesias in both lower limbs, decreased walking distance, and occasional urinary incontinence. His medical history included thrombophlebitis of the lower limbs in 2009 and glaucoma in the left eye, diagnosed in 2017. The patient's preoperative clinical examination revealed a strength deficit of 4/5 in the right L5 dermatome, and both Lasègue and Wassermann signs were positive bilaterally, indicating significant nerve root compression. The patient's general condition was stable, and his vital signs were within normal ranges prior to surgery. A lumbar MRI was performed, which revealed protrusive posterior median disc herniations at the L3-L4 and L4-L5 levels, causing significant compression of the nerve roots and the dural sac. In addition, there was substantial disc space narrowing and inflammation of the vertebral endplates from L3 to S1. Based on these imaging findings, the patient was diagnosed with lumbar spinal stenosis and degenerative disc disease. The decision was made to proceed with a TLIF procedure to decompress the nerve roots and stabilize the affected vertebral segments .
|
Neurosurgery
|
The surgery was performed under general anesthesia, with the patient positioned prone on the operating table to allow optimal access to the lumbar spine at HJ Hospitals, Limete, Kinshasa. Standard TLIF techniques were employed, including the placement of interbody cages and pedicle screws at the L3-L4 and L4-L5 levels. Intraoperative neuromonitoring was used to ensure the preservation of nerve function throughout the procedure. However, a critical issue with head positioning occurred during the surgery. The custom-made round headrest, designed to support the patient's face and protect the eyes from compression, was improperly positioned. This resulted in prolonged bilateral ocular compression, particularly affecting the patient's left eye, which had a pre-existing glaucoma diagnosis. Although the surgery itself proceeded without complications and the desired lumbar stabilization was achieved, the intraoperative head positioning error was not recognized until the patient awoke in the post-anesthesia care unit.
|
Neurosurgery
|
Upon awakening, the patient reported complete vision loss in his left eye, accompanied by persistent tearing from the affected eye whenever he attempted to focus his vision. Ophthalmological consultation was urgently requested, and a comprehensive examination revealed that the patient's glaucoma had decompensated, likely due to the prolonged external compression of the eye during surgery. IOP was significantly elevated in the left eye, and optic nerve damage was suspected. Initial medical management included the use of glaucoma eye drops to reduce intraocular pressure and prevent further damage to the optic nerve. However, after three weeks of observation, there was no improvement in the patient's vision, prompting the decision to perform glaucoma surgery. Unfortunately, despite this intervention, the patient's vision remained severely compromised, with complete blurring of vision in the left eye persisting at the three-month follow-up.
|
Ophthalmology
|
The patient's vision in the left eye, which had been stable and functional prior to surgery, was permanently affected by the intraoperative complication. At the three-month follow-up, ophthalmological assessments confirmed that the optic nerve damage was irreversible, and the patient's vision in the left eye remained completely blurred. This complication significantly impacted the patient's postoperative recovery and overall quality of life, as the vision loss in one eye affected his daily activities and independence.
|
Ophthalmology
|
Despite the severe ophthalmological complication, the patient's spinal surgery achieved its intended goals. The TLIF procedure successfully decompressed the nerve roots at L3-L4 and L4-L5, alleviating the patient's lumbar radicular symptoms. At the three-month postoperative follow-up, the patient reported significant improvement in pain and mobility, with no residual neurological deficits in the lower limbs. The surgical site healed well, and there were no signs of infection or hardware failure. The patient was able to gradually resume normal activities with physical rehabilitation, although the visual impairment continued to limit his full recovery .
|
Neurosurgery
|
The management of POVL requires timely diagnosis and intervention to prevent further deterioration of vision. In the immediate postoperative period, any patient reporting visual changes should undergo an urgent ophthalmologic evaluation. In our case, the patient developed complete vision loss in the left eye immediately after the TLIF procedure, and an ophthalmology consultation was sought. Despite initial attempts to manage the condition medically with topical glaucoma medications, there was no significant improvement, and the patient ultimately required glaucoma surgery .
|
Ophthalmology
|
This case demonstrates the importance of comprehensive preoperative screening for patients with known ocular conditions such as glaucoma. A detailed ophthalmologic assessment, including IOP measurements and optic nerve evaluations, should be part of the routine preoperative workup for patients undergoing prone-positioned surgeries. Identifying patients at increased risk for ocular complications allows for more targeted interventions, such as modified positioning strategies or intraoperative monitoring of IOP, to reduce the likelihood of vision loss.
|
Ophthalmology
|
This case highlights the rare but serious complication of POVL following TLIF, specifically due to glaucoma decompensation triggered by improper intraoperative positioning. Although TLIF is a widely utilized and generally successful procedure for managing lumbar degenerative conditions, this case underscores the critical importance of paying meticulous attention to patient positioning, particularly in prone surgeries where ocular compression is a risk factor. While POVL is a rare complication, its potential to cause permanent and life-altering outcomes necessitates a proactive and preventive approach. This case serves as a stark reminder that even the most routine of procedures carries risks, and it is the responsibility of every surgical team to minimize those risks through careful planning, vigilance, and adherence to best practices. By learning from this case and implementing targeted preventive strategies, we can improve the safety of spinal surgeries and reduce the incidence of POVL, ensuring better outcomes for patients undergoing these complex procedures.
|
Neurosurgery
|
One of these 11 patients (P9) was the most severe case, with severe congenital hypotonia, distal malformations (club feet, arachnodactyly), dysmorphic features (microretrognathia, ogival palate), facial weakness, and suction and swallowing difficulties requiring a gastrostomy. The patient developed spinal stiffness, periscapular amyotrophy, and pes cavus. The patient was weaker in the upper limbs, was able to bend arms against gravity, but had forearm extensor weakness with drooping hands. The evolution of the pathology was favorable: gastrostomy was closed before 10 years and nocturnal ventilation was stopped in early childhood.
|
Neurology
|
All four patients presented with hypotonia in the neonatal period and showed motor weakness, but did not show joint contractures or scoliosis. P15 and P16 showed a favorable course. P15 was able to walk without too much delay, climb stairs soon afterwards during infancy, and run during childhood, with a slight weakness of the waist and proximal part. P16’s condition was more severe, and she was wheelchair-bound during childhood, started to walk with assistance during adolescence, and walked without assistance indoors at adulthood. Muscle atrophy was predominant on the posterior thigh and the anterior leg during her last consultation.
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Neurology
|
Additionally, free gingival grafts are good indications for the StellaLife Recovery kit, due to the healing of secondary intention at both the donor and recipient sites. Patients have reported a therapeutic effect in our office when placing the gel over the donor site on the palate. Figure 14 demonstrates a patient referred for inadequately attached gingiva #20 (American tooth numbering system; FDI #35). The patient elected to have a free gingival graft rather than connective tissue graft after discussing both options. The recipient was bed-prepared utilizing a #15 blade. Epithelialized tissue was harvested from the patient’s palate and fixated to the recipient site utilizing periacryl oral tissue adhesive . The graft was immobilized before the patient was released. At 2 weeks post-operatively, accelerated would healing was observed with no inflammation present .
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Periodontology
|
In this article, we present the case of an 87-year-old male with aCRC carrying CCDC6::CASP7 rearrangements, caused by an inversion of the intergenic segment between the two genes. In addition to fusion, somatic mutations of proto-oncogenes PIK3CA (phosphoinositide-3-kinase catalytic subunit alpha) ( E542K ) and KRAS (kirsten rat sarcoma viral oncogene homolog) (G12C) were detected ( Supplementary Materials ). The patient underwent surgical resection but died from fecal peritonitis after the operation.
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Oncology
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An 87-year-old man was referred by his physician for abdominal pain of months of evolution and recent vomiting. Computed tomography (CT) imaging showed a perforated cecum neoplasm with hepatic metastases. Immediate right hemicolectomy was performed in May 2019, in the Surgery Department of Hospital Nuestra Señora de Sonsoles (Ávila, Spain). During surgery, an enlarged tumor was observed that reached the wall of the right iliac fossa and featured abundant purulent and fecaloid liquid in the abdominal cavity. The pathology department received a 26 cm segment of the right colon with a perforated area. Pathological findings revealed a moderately differentiated adenocarcinoma with microsatellite stability (MSS). It was microscopically confirmed that the tumor infiltrates serosa (pT4a) with metastasis in 7 out of 16 lymph nodes analyzed (pN2b).
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Oncology
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The donor was a 34-year-old brain-dead female who suffered a subarachnoid hemorrhage. She had a mild smoking history (5 packs/year) and was located in a hospital on an Italian island more than 1300 km away from our center, with an estimated transport time of more than 6 h. The donor had been hospitalized in the ICU for 2 days prior to procurement. The organs were initially judged to be low-quality due to the presence of abundant fluid secretions at the bronchoscopy upon arrival at the hospital, a low PO 2 /FiO 2 (P/F) ratio of 290 mmHg at the moment of the organ offer, and the presence of edema and contusions from cardiopulmonary resuscitation maneuvers revealed at the retrieval, resulting in an OTO score of 9 . The recipient was a 48-year-old man suffering from chronic respiratory failure related to hypersensitivity pneumonitis.
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Pulmonology
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At the first functional assessment, the P/F ratio was 366 mmHg, vascular resistances were 1024 dyn.s/cm 5 , and the mean pulmonary arterial pressure was 5 mmHg. Upon visual inspection, the lungs were still mildly edematous with heavy bases and moderate foamy secretions detected at the first bronchoscopy check.
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Pulmonology
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A 78-year-old female (weight 72 Kg; height 170 cm) was admitted with retrosternal pain, of moderate intensity, without relief or aggravating factors, associated with nausea, vomiting, and dysphagia for liquids and solids. No other symptomatology was found. The patient had a history of controlled arterial hypertension, first-degree atrioventricular (AV) block, and hypothyroidism. Her current medications included alprazolam 0,5 mg PO once daily, levothyroxine 0.125 mg PO once daily, and lorazepam 1 mg PO at bedtime and she was allergic to fosfomycin. The airway evaluation showed a Mallampati I without any signs of a potentially difficult airway. Standard preoperative fasting was ensured.
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Anesthesiology
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A 12-lead electrocardiogram showed a sinus rhythm, 75 bpm, compatible with first-degree AV block and occasional premature ventricular beat. High-sensitivity troponin levels turned out normal. The initial chest X-ray revealed an intrathoracic gastric air-fluid level , which was confirmed by a CT scan : a large air-fluid level within the left hemithorax, exerting pressure on the left lung and resulting in a mediastinal shift toward the right side, along with deviation of the trachea and heart.
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Cardiology
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The patient was premedicated with an intravenous proton pump inhibitor. Upon arrival to the operation room, the patient was fully conscious and cooperative and complained of mild chest discomfort. Standard ASA monitoring was used, and radial artery catheterization was performed for continuous invasive blood pressure. Heart rate (HR) was 75 beats/min, blood pressure (BP) was 110/62 mmHg, and pulse oximetry (SpO 2 ) was 89% with a high concentration mask.
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Anesthesiology
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The attempt at endoscopic decompression of the stomach and subsequent laparoscopic surgical reduction was unsuccessful. Therefore, the decision was to proceed to a midline laparotomy with a reduction of the entire hiatal hernia and retraction of the stomach into the abdominal cavity, which later revealed signs of distress. A chest drain was placed on the left side. Occasional premature ventricular beats, followed by ventricular tachycardia, were observed during hernia repair and ceased after the interruption of the maneuver.
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Surgery
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After being followed for up to three months, the patient was free of thoracic or abdominal discomfort, reflux, vomiting, or symptoms of heart failure. Meanwhile, the re-evaluation thoracic CT scan showed signs of previous surgical repair of a left diaphragmatic hernia, with moderate elevation of the left diaphragmatic hemi-dome and sliding esophageal hiatal hernia, with part of the stomach intra-thoracic .
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Gastroenterology
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The presented clinical case highlights the management of an intrathoracic full stomach with ATI to prevent potential respiratory complications. Intrathoracic stomach, resulting from a hiatal hernia, is a rare but significant condition. The abnormal positioning of the stomach within the thoracic cavity could possibly cause complications such as lung compression, resulting in respiratory distress. Associated symptoms such as dyspnea and chest pain or arrhythmia, including sinus tachycardia, atrial flutter, atrial fibrillation, extrasystole, and even cardiac arrest, are possible due to compression of the heart . Immediate surgical intervention is often warranted to alleviate these symptoms and prevent further complications. The decision to proceed with urgent surgical repair was based on the patient's condition and the need for prompt intervention.
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Gastroenterology and Thoracic Surgery
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This case of an intrathoracic full stomach, proposed for urgent surgical repair, highlights the significance of conscious intubation as a valuable technique in preventing respiratory complications and allows for timely intervention while maintaining the patient's cooperation. However, careful consideration of patient factors, collaboration between the surgical and anesthesia teams, and individualized decision-making are essential in determining the most appropriate approach for each patient.
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Anesthesiology
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A nine-month-old male patient was diagnosed with SCID following hospitalization due to Pneumocystis jirovecii pneumonia. Genetic testing revealed a homozygous mutation of the CD3D gene associated with the T-B+NK+ SCID phenotype. Subsequently, at 13 months old, he underwent an unrelated umbilical cord blood transplant (five of six human leukocyte antigen (HLA)-matched alleles). The conditioning regimen consisted of busulfan, 90 mg/h/L, fludarabine, 160 mg/m 2 , and anti-thymocyte globulin (ATG), 10 mg/kg.
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Immunology
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Eight months after the transplant, he was admitted to the hospital due to fever with no apparent focus of infection and new-onset hemolytic anemia (minimum value of hemoglobin 3.3 g/dL). Laboratory findings supporting hemolysis included elevated serum lactate dehydrogenase (LDH) and bilirubin, reticulocytosis, and decreased haptoglobin. A direct antiglobulin test (DAT) was positive, with IgG and C3 antibodies found bound to red blood cells, which combined with elevated hemolysis parameters was consistent with the diagnosis of autoimmune hemolytic anemia. He received two red blood cell-compatible transfusions and was treated with methylprednisolone 30 mg/kg per day for three days and intravenous immunoglobulin (IVIG) 1 g/kg. After the increase in hemoglobin levels, the patient was discharged on prednisolone 1.5 mg/kg/day and maintained regular outpatient follow-up appointments (Table 1 ).
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Hematology
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Approximately one year after the diagnosis of AIHA, and while still on corticoid treatment (prednisolone 0.5 mg/kg/day), he had a symptomatic hemolytic relapse. Hemoglobin levels decreased to 8 g/dL. Therapy was escalated, and he was treated once weekly with rituximab 375 mg/m 2 for a total of four doses, maintaining treatment with prednisolone (Table 1 ). There was an excellent response to treatment with a considerable improvement in hemoglobin levels (14.8 g/dL), normal haptoglobin levels (127 mg/dL), and a decrease in LDH and bilirubin (now 0.3 mg/dL and previously to therapy at 1.94 mg/dL). After continued stabilization of hemoglobin levels, prednisolone was gradually and slowly tapered until complete suspension was achieved, four months after treatment with rituximab. Monitoring after therapy with rituximab revealed maintained normal levels of immunoglobulins, with no need for substitution therapy. No infectious complications occurred.
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Hematology
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Four months after corticosteroids were discontinued, the patient presented with fatigue, abdominal pain, and jaundice. Laboratory evaluation showed hemoglobin 6.3 g/dL, absolute reticulocyte count 784x10^9/L, total bilirubin 2.85 mg/dL, direct bilirubin 0.22 mg/dl, and LDH 1178 U/L, confirming another hemolytic crisis. No concomitant cytopenias were present. Positive DAT for IgG and C3 supported the diagnosis of warm AIHA. Second-line treatment with mycophenolate mofetil in association with prednisolone was initiated with a favorable hematologic response. A month later, a slow reduction in corticoid dosage was attempted and tolerated. Shortly after, corticosteroids were discontinued with success. The patient, now five years old, maintains monotherapy with mycophenolate mofetil (600 mg/m 2 /dose twice daily) and has not experienced any further relapses (Table 1 ). His hemoglobin level at the last visit was 12.3 g/dL, and his last follow-up immune workup revealed T lymphocyte subsets , B lymphocyte subsets (651/uL), natural killer cells (328/uL), and normal immunoglobulin levels for age.
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Hematology
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A 13-year-old male patient with a medical history of aplastic anemia underwent allogeneic HSCT from an HLA-matched sibling donor (10/10 HLA matches). The conditioning regimen administered consisted of cyclophosphamide and ATG. Graft-versus-host disease prophylaxis was carried out with cyclosporine and methotrexate.
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Hematology
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In the weeks following the transplant, the child developed several complications, including necrotizing fasciitis with septic shock. Pseudomonas aeruginosa was isolated, and he was treated with broad-spectrum antibiotics and surgical debridement. Three months later, following reconstructive surgery with skin grafting, he presented with fever along with a progressive reduction in hemoglobin (minimum value of hemoglobin 3.0 g/dL) and platelet levels (minimum platelet count of 5×10^9/L). An initial evaluation for hemolysis showed elevated LDH and bilirubin. The DAT was positive. Autoimmune bicytopenia possibly accompanied by microangiopathic hemolytic anemia secondary to cyclosporine was suspected at this point. Cyclosporine was discontinued, and the patient started treatment with corticosteroids (methylprednisolone 30 mg/kg for three days), IVIG (1 g/kg), and plasmapheresis (Table 1 ). Supportive care with red blood cell concentrates and fresh frozen plasma was provided. The patient failed to respond to the described therapy, and, after a multidisciplinary discussion, he was proposed for treatment with weekly administrations of rituximab 375 mg/m 2 . After the second administration, there was a progressive improvement in hematological parameters, and after one week, hemoglobin rose to 9.9 g/dl and platelet count to 75×10^9/L. Taking into account the favorable hematological response and in order not to exacerbate immunosuppression, it was decided to suspend rituximab after the first two administrations. No substitution therapy was needed, and no infectious complications occurred.
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Hematology
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One month later, the patient experienced a relapse. Laboratory evaluation at admission showed hemoglobin of 6.0 g/dL, platelet count of 8X10^9/L, total bilirubin of 3.82 mg/dL, LDH of 682 U/L, and haptoglobin lower than 10 mg/dL. The possibility of transplant-associated thrombotic microangiopathy was investigated, but ADAMTS13 activity was normal. The DAT remained positive, with IgG and C3 antibodies found bound to red blood cells, which was consistent with the previous diagnosis of autoimmune cytopenias. Treatment with corticosteroids and immunoglobulin was started, and he completed four additional doses of rituximab. Despite these treatments, there was an initial decrease in hemoglobin (minimum value of 3.0 g/dL). Supportive care with red blood cell concentrates and fresh frozen plasma was again administered as needed in the acute phase.
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Hematology
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Considering the failure to respond to the previously prescribed therapeutics for AIHA and autoimmune thrombocytopenia, he initiated treatment with mycophenolate mofetil in association with prednisolone (1 mg/kg) (Table 1 ). After starting this new line of treatment, there was a satisfactory and consistent response with sustained normal levels of hemoglobin and platelets. The patient, now 18 years old, maintains regular follow-up appointments. He has been gradually reducing dosages over the years, without further relapses. Recently, it was possible to suspend both mycophenolate mofetil and prednisolone, and he has been stable since. On his last visit, he had a hemoglobin level of 15.0 g/dL (Table 1 ).
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Hematology
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A four-month-old boy with no prior medical history presented at the hospital with three days of fever along with irritability and skin pallor. Laboratory evaluation revealed bicytopenia (anemia and thrombocytopenia), elevated C-reactive protein and liver enzyme levels, as well as slight coagulation abnormalities. Physical examination also highlighted a significant splenomegaly and light silvery-colored hair. Despite early initiation of antibiotic therapy with ceftriaxone, there was a progressive clinical and laboratory deterioration. Hemophagocytic lymphohistiocytosis (HLH) was suspected, and additional bloodwork was ordered. Ferritin levels of 30.000 ng/mL and CD25 levels of 47.000 IU/mL, in addition to the patient’s symptoms, met the clinical and laboratory criteria for HLH, confirming the diagnosis.
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Hematology
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After clinical stabilization, the patient was evaluated by a multidisciplinary team. Partial albinism with hypopigmentation of the skin and hair, associated with HLH, raised suspicion for Griscelli syndrome. Therapy for HLH was initiated with methylprednisolone (2 mg/kg/day), cyclosporine (2 mg/kg/day, aiming at serum levels around 200 ng/ml), and alemtuzumab (1 mg/kg/day) with a good clinical response. Genetic testing confirmed the diagnosis of Griscelli syndrome, identifying a heterozygous mutation in the RAB27A gene (c.99A>G and c.467+1G>c). The patient was subsequently proposed for allogeneic HSCT from an unrelated donor (10/10 HLA compatibility), which was performed about a month later. The conditioning regimen consisted of busulfan, 90 mg/h/L, fludarabine, 160 mg/m 2 , and alentuzumab, 1 mg/kg (Table 1 ).
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Hematology
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Approximately six months after HSCT, the patient presented at one of the regular evaluations with increased cutaneous pallor and asthenia. Analytical evaluation revealed a decrease in hemoglobin levels to 7.7 g/dL associated with elevated hemolysis parameters (elevated LDH and bilirubin, and low haptoglobin). The DAT was positive for IgG1 and C3d, confirming the diagnosis of AIHA. Therapy with prednisolone (6 mg/kg/day) was initiated but with only a partial response. After a new multidisciplinary meeting, it was decided to add rituximab to the therapy in four weekly doses of 375 mg/m 2 . The patient had an excellent response to therapy, with an increase in hemoglobin levels to 12.4 g/dL and normalization of hemolysis parameters. It was possible to gradually reduce corticosteroid dosage, which was discontinued two months after starting rituximab therapy.
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Hematology
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About three months after discontinuation of corticosteroids, a relapse of AIHA was noted, with a decrease in hemoglobin levels to 5.4 g/dL and a new elevation of hemolysis parameters. The DAT remained positive. Therapy with prednisolone was resumed, but re-initiation of corticosteroid therapy was insufficient to control AIHA, so four more doses of rituximab 375 mg/m 2 were administered once a week with a favorable clinical and hematological response (increase in hemoglobin to 12.3 g/dL). The patient, currently 22 months old, is still undergoing a slow and gradual corticosteroid taper, maintaining stable hemoglobin levels. On his last visit, he had a hemoglobin level of 13.0 g/dL (Table 1 ).
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Hematology
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A previously well 18-year-old female of Indian descent presented with a 2-year history of recurrent right foot blisters after minor trauma. The painful tense blisters were confined to the distal right lower limb. She had received multiple courses of antibiotics to poor effect. On examination, a single tense bulla with hyperpigmented rim was seen on the right lateral malleolus . Previous blister sites healed as stellate atrophic scars with peripheral hyperpigmentation . A punch biopsy was performed, and histological findings are shown below . Direct immunofluorescence revealed heavy fibrin exudates within the blood vessels. Fig 1 Fig 2
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Dermatology
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A 70-year-old female suffering from low back pain with right radicular pain since 2016. While lumbar epidural injection provided relief of lower extremity symptoms, the patient had ongoing low back pain radiating to both hips. The patient received bilateral L3-L5 MB RFA using the parasagittal and sub-mammillary technique. Fourteen months following the procedure, the patient elected to have repeat RFA treatment while self-reporting “excellent” (90 %) pain relief for 13 months and ongoing partial (75 %) pain relief.
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Pain management
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A 54-year-old male patient suffering from low back pain following a motor vehicle collision received bilateral L3-L5 MB denervation. Following the parasagittal and sub-mammillary technique, the patient self-reported excellent (90 %) pain relief for 11 months. At 12 months, the patient had ongoing partial (10–20 %) pain relief and elected to have repeat RFA treatment. The patient stated this was the “best result to date” from RF ablation in terms of quality of pain relief.
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Pain management
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A female 78-year-old patient received L3-L5 MB denervation on the right side using the parasagittal and sub-mammillary technique. At 15 months following RFA treatment the patient self-reported complete (100 %) pain relief that is ongoing with no recurrence of symptoms. The patient stated this was the “best result ever” from any previous RFA and elected to delay repeat treatment.
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Pain management
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A 75-year-old female was treated with bilateral L3-L5 MB denervation using the parasagittal and sub-mammillary technique. At the 17 months post-procedure appointment, the patient self-reported partial pain relief (80 %) for 16 months and 1 month of ongoing relief at 25 %. The patient stated this was the “best result of any ablation” previously received.
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Pain management
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Therefore, a coronary angiography is performed to confirm the pathological Cardiac CT scan in November 2020 and shows significant LAD1 stenosis and moderate downstream atheroma; vascular theft on the left Circumflex artery, no opacification, voluminous Circumflex fistula heading towards the right atrium; 40 % distal right coronary artery stenosis . Fig. 4 Fistula on coronary angiography. Fig. 4 Fig. 5 Fistula on coronary angiography. Fig. 5
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Cardiology
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Because of a coronary artery disease that has become significant and is considered as a multivessel coronary disease but also due to the patient's good general condition and active lifestyle and the giant size of the fistula, cardiac surgery is decided with two aims: aorto-coronary bypass and fistula closure. The procedure is as follows: distal AMIG-IVA bypass and ligation of the coronaro-cava fistula from the Circumflex artery . Surgery is a success. Subsequently, a minimal right-sided pericardial effusion was noted without clinical impact. The patient then underwent a recovery period in cardiovascular rehabilitation, achieving complete recovery of autonomy and usual physical activity. The echocardiogram at 6 months post-surgery was strictly normal. A stress test was performed one year after the surgery and only showed non-specific inferolateral repolarization abnormalities. Fig. 6 Fistula's ligation surgery. Fig. 6
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Cardiology
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A 58-year-old female presented to neurosurgery with severe right-sided facial, tongue, and jaw pain unresponsive to multiple conservative therapies including medication. T2-weighted MRI with FIESTA sequence demonstrated right PICA compression of the right glossopharyngeal nerve entry zone .
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Neurosurgery
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A retrosigmoid craniotomy for decompression of Cranial Nerves IX and X was performed. After opening the dura, microscopic visualization was used to open the cerebellopontine angle cistern and identify Cranial Nerves VII, IX, X, and XII. PICA was also identified. After failure to stimulate at 0.2 mA, Cranial Nerve IX was divided to visualize the root entry zone of Cranial Nerve X. Pledgets were placed between the PICA and the entry zones of Cranial Nerves IX and X before closure ( Supplementary Video 1 ). Patient obtained significant relief of preoperative pain symptoms without complication and was discharged home with routine follow-up planned on postoperative Day 2.
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Neurosurgery
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This case report describes the first experience of incorporating AR into the presurgical planning of an MVD for glossopharyngeal neuralgia. While we describe the notable value of the system, we did not directly compare surgeries performed with and without AR. Future studies drawing these comparisons are warranted. Here we describe the use of an augmented reality system to aid in planning an MVD for glossopharyngeal neuralgia. The system provided the care team with a heightened understanding of the patient’s anatomy, and the ability to register the 3D models with the patient’s head was felt to be valuable in positioning, pre-surgical visualization, and incision planning.
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Neurosurgery
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An 85-year-old male was referred to a colorectal surgeon with an appendiceal nodule bulging into the caecum on routine colonoscopy . A considerably smaller protuberance was noted 5 years prior with a plan for surveillance, he was asymptomatic to the appendiceal nodule and otherwise only reported dyspeptic symptoms. He has a background of peptic ulcers, obstructive sleep apnoea, hypertension and laparoscopic cholecystectomy. He has no known history of genetic syndromes. A CT abdomen-pelvis was organized to further define the appendiceal pathology. The appendix was of normal calibre, without definite nodular thickening and comparable to a previous study in 2019. Further there was no evidence of lymphadenopathy or metastatic disease.
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Gastroenterology
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Due to the interval increase in size on colonoscopy, the patient proceeded to a laparoscopic stapled caecotomy, for which he recovered well and was discharged Day 1 post-operatively. The histopathology report found a benign neural tumour bulging into the appendiceal orifice and obliterating the lumen . The tumour was located in the base of the appendix, 15 mm from the resection margin. It contained Schwann cells admixed with mast cells and ganglion cells which stained positive for S100 . The findings were consistent with a benign mature GN.
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Pathology
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A 24-year-old Indian man, who has been living in Lisbon, Portugal, for three years and works for a company installing air conditioning, presented to the Emergency Department with three days of left-sided pleuritic chest pain radiating to the back. He had no relevant medical or surgical history and denied associated symptoms, such as dyspnea, cough, or fever. Clinical examination revealed decreased breath sounds at the left lung base on pulmonary auscultation. Chest radiography showed a nonspecific round opacity in the lower lobe of the left lung. A chest computed tomography (CT) scan was performed, revealing a unilocular cystic nodule measuring approximately 72 x 46 mm. The scan was then extended to the abdominopelvic region, identifying another cystic nodular formation (63 x 38 mm) with slightly thickened walls in the left liver lobe, consistent with a hydatid cyst . A cranial CT scan showed no lesions. Laboratory findings highlighted a C-reactive protein level of 97.5 mg/L and positive anti- E. granulosus antibodies with a titer of 1/640. The remaining blood tests were normal, and other serological studies were negative. He was started on albendazole 400 mg every 12 hours. A cranial magnetic resonance imaging (MRI) and a thoraco-abdominopelvic MRI confirmed the findings described in the CT scans. Two weeks later, an elective laparoscopic resection of hepatic segments II/III was performed, removing a 9 x 6 x 5 cm lesion, histologically consistent with a hydatid cyst . He continued albendazole therapy and underwent a left lower lobectomy two months later, which was also confirmed histologically as a hydatid cyst. The patient was discharged without complications and instructed to continue albendazole therapy until the next evaluation but was subsequently lost to follow-up after immigrating to Switzerland.
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Parasitology
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This case highlights the importance of considering the epidemiological context to enable early diagnosis and treatment, leading to successful surgical management and extraction of hydatid cysts from the lung and liver. Therefore, in a patient originally from or previously residing in an endemic area, or with a possible history of exposure, who presents with respiratory or abdominal symptoms - especially when cysts are identified - echinococcosis should be considered a potential diagnosis. In most cases, including our patient's case, surgery combined with anti-parasitic therapy is generally the definitive treatment, aiming to prevent complications that can lead to larval seeding. While surgical intervention can be curative, cyst recurrence is common in the absence of anti-parasitic treatment. Patients are advised to undergo serial imaging over several years post-resection to monitor for refractory seeding and recurrence. This case underscores the significant risk associated with lack of follow-up, which may increase the likelihood of undetected recurrence and related complications .
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Parasitology
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This case highlights the importance of consideration of non-malignant diagnoses mimicking upper tract urothelial carcinoma (UTUC), and using a structured approach to diagnosis.
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Urology
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A male in his 50s presented to the Emergency Department with right-sided colicky abdominal pain, vomiting, haematuria, and reduced urinary output. He was normally well, with a past medical history of gout, hypertension, and polyarthritis. There was no history of sexually transmitted infections. He was a non-smoker and did not drink alcohol. His vital observations were stable on admission. On examination, he was tender in the right flank; however, there was no guarding or peritonism. A digital rectal examination revealed no obvious abnormalities. Blood testing on admission showed a normal white cell count but a significantly raised C-reactive protein, urea, creatinine, and potassium (Table 1 ). No recent baseline bloods were available. A urine screen was positive for leucocytosis and nitrites; however, cultures revealed no significant growth. Blood cultures were also negative.
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Urology/Nephrology
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The patient had a urethral catheter inserted and was commenced on broad-spectrum intravenous antibiotics. Non-contrast computed tomography (NCCT) of the urinary tract, completed soon after admission, showed a large soft tissue lesion at the level of the distal third of the right ureter, measuring 2.6 cm by 2.4 cm by 2.8 cm, resulting in moderate hydroureteronephrosis and concerning for a UTUC . Left renal atrophy was noted, and therefore, urgent decompression of the right kidney was undertaken.
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Urology
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Given the above findings, urgent drainage of the right kidney was achieved via the insertion of a nephrostomy tube, rather than a ureteric stent, owing to the urgency of the case and the availability of operating theatres and interventional radiology. As a result, the patient’s renal function steadily improved. An interval CT urogram and thorax scan showed a reduction in hydroureteronephrosis of the right renal collecting system and a persisting obstructing mass at the distal right ureter, with perinephric inflammatory change. There was no evidence of metastatic disease. With a working diagnosis of UTUC, the case was discussed at the uro-oncology multi-disciplinary meeting (MDM), which recommended urine cytology. The plan was to proceed to the right distal ureterectomy if the result was positive, and ureteroscopy if negative. Three urine samples from the nephrostomy tube were sent for cytology, all of which came back negative for malignancy. Renal function took a number of days to recover; creatinine halved from 670 µmol/L to 325 µmol/L in five days and was back to baseline (120 µmol/L) in one month's time. C-reactive protein normalised in two weeks. An outpatient cystoscopy and ureteroscopy revealed no abnormalities. Following this, a magnetic resonance imaging (MRI) of the abdomen was completed, which revealed asymmetry of the right seminal vesicle with hyperintensity on diffusion-weighted imaging. It also reported that no solid mass lesion was associated with the right distal ureter, and there was no evidence of metastatic disease. A renal dimercaptosuccinic acid (DMSA) scan showed a left atrophic kidney with differential uptake of 15%, and 85% in the right kidney.
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Urology
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This case report presents a rare instance of seminal vesiculitis leading to unilateral ureteric obstruction, a condition that mimicked UTUC in its presentation. This underscores the challenges of diagnosing seminal vesiculitis, as it is a rare condition with symptoms that can overlap with other urological diseases. Diagnostic imaging and a structured approach to exclude malignancy proved crucial in this case, and MRI, rather than CT, provided the definitive diagnosis, suggesting its importance in complex cases of obstructive uropathy.
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Urology
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This case emphasises the need for clinicians to consider non-malignant cases of ureteric obstruction, particularly in those who present atypically, and to employ appropriate diagnostic techniques, such as MRI and rectal examination, to secure an accurate diagnosis. I believe this case highlights the importance of obtaining a definitive diagnosis in patients with suspected UTUC before proceeding to radical treatment.
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Urology
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A73-year-old female with no history of smoking was admitted to the hospital on August 13, 2020, with shortness of breath for more than 1 month. More than 1 month ago, the patient developed shortness of breath when she was in the right decubitus position, but the left decubitus position improved. During deep inhalation, she heard a high aspiratory laryngeal sound with no difficulty eating, chest tightness, chest pain, nausea, or vomiting. The patient was not affected by life and did not seek medical treatment. Physical examination revealed a pulse of 74 beats/min, breathing of 22 beats/min, blood pressure of 124/65 mm Hg, body temperature of 37°C, clear consciousness, no cyanosis of the lips, no irritation of the jugular vein, no swelling of the superficial cervical lymph nodes, centered trachea, triscuval sign on aspiration, and clear breath sounds in both the lungs. She had a history of radiotherapy for a nasopharyngeal malignant tumor 16 years prior, no recurrence during long-term follow-up, and type II diabetes for several years. CT examination of the neck and chest revealed soft tissue shadow with calcification on the left posterior wall of the initial tracheal segment, 1.6 × 1.0 cm in size and 2.2 cm in length, infiltrating the wall of the trachea near the whole layer, with local tracheal cavity stenosis accounting for about 80% , and there were no significantly enlarged lymph nodes. Serological examination of the tumor indicated that carcinoembryonic antigen, CA125, squamous cell carcinoma-associated antigen, and alpha-fetoprotein (3rd generation) levels were within the normal ranges. On August 14, 2020, a painless bronchoscopic airway neobiological biopsy was performed, and it was observed that the glottis was well closed, new organisms were present below the glottis, the lumen was narrow, and the mirror barely entered. In this biopsy, the lower trachea was unobstructed, the carina was sharp and centered, and the mucosa was normal.
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Pulmonology
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Treatment process: After the diagnosis, a multidisciplinary joint diagnosis and treatment discussion was conducted under the auspices of the respiratory department of a superior hospital. As the patient had a history of head and neck radiotherapy for nasopharyngeal cancer, her general condition was poor, and her body mass index was 16.4, she could not undergo total resection or chemotherapy. At the same time, the patient had obvious shortness of breath, which needed to be treated as soon as possible to improve the clinical symptoms and prolong the life of the patient. Therefore, partial tumor resection was performed under general anesthesia with tracheoscopy followed by local radiotherapy. Gross tumor volume: 60GY/25Fx, planning tumor volume: 50GY/25Fx, and radiotherapy was performed 20 times, with sudden shortness of breath. CT of the neck revealed a local tracheal stenosis involving the subglottis. Consultation with the otolaryngology department was requested, and emergency tracheotomy was performed on October 9, 2019. As the tumor could not be completely cured, the patient experienced repeated coughing and shortness of breath and died of the disease 15 months later.
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Oncology
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We report a case of cutaneous melioidosis that describes the first clinical isolate of B. pseudomallei from Australia that exhibits meropenem resistance without prior exposure.
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Infectious diseases
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In November 2021, a 54-year-old male with type II diabetes mellitus, chronic kidney disease, polyarthropathy of unclear cause on regular prednisolone (12 mg daily) and metabolic syndrome presented to the hospital with multiple cutaneous abscesses on the right lower quadrant of the abdomen and right thigh. These had started on the abdominal wall in May 2021 after he had been walking through river water with open skin wounds. His local community doctor had prescribed multiple short courses of oral antibiotics, which included doxycycline and trimethoprim–sulfamethoxazole. The patient denied previous meropenem exposure. The lesions initially improved with trimethoprim–sulfamethoxazole and doxycycline; however, post-cessation, there was a re-accumulation of pus. Upon further history and examination, no systemic symptoms were identified. The patient underwent surgical debridement of the abdominal wall lesions. A surgical tissue specimen was processed in the microbiology laboratory and set up on horse blood agar (HBA) (both in 5% CO2 and anaerobic incubation conditions), MacConkey and crystal violet, colistin and nalidixc acid and chocolate agar (CHOC). Growth was monomicrobial and pure. The isolate was culture positive for B. pseudomallei .
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Infectious diseases
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We present a patient with an unusually painful, multiloculated acromioclavicular joint cyst, with initial imaging findings concerning for soft tissue sarcoma, who was treated with excisional biopsy of the cyst and resection of her distal clavicle.
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Orthopedic surgery
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A 73-year-old female with history of gout, rheumatoid arthritis, right carpal tunnel, and right hip and left knee arthroplasties presented with a slowly enlarging mass in the area above her left shoulder that was present for several years duration. She had not previously undergone biopsy or any cross-sectional imaging and was referred to surgical oncology for further evaluation and to rule out soft tissue sarcoma as she was concerned about a possible tumor. She denied any recent or remote history of trauma to the area. She endorsed significant pain in the area of the mass. On physical exam, there was a palpable, soft, mobile mass on top of her left shoulder, roughly 5 cm in diameter. The mass was extremely tender to palpation. She had full elbow, wrist, and hand range of motion. Sensation was intact distally. There was no clinical or laboratory evidence of infection.
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Oncology
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The patient underwent en bloc resection of cyst containing portion of trapezius, distal clavicle, and AC joint. The specimen was sent for permanent section. Resection of 0.5 cm of the distal clavicle and AC joint was then performed. Her rotator cuff tear was found to be irreparable and given that it was minimally symptomatic, no cuff repair was pursued. A Blake drain was placed in the resection defect and soft tissue closure was performed. Postoperatively, she was placed in a sling for comfort as needed.
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Orthopedic surgery
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In our described case, the patient was concerned about the appearance of the mass and the pain it caused her. Her cuff-tear arthropathy was well compensated and minimally symptomatic. We elected to pursue distal clavicle excision due to the significant size of the cyst, irreparable and asymptomatic nature of her rotator cuff tear, and concern for possible neoplastic etiology. This method also allowed for wide excision of the lesion in the event that surgical pathology was consistent with malignancy. Similar approaches to address AC joint cysts without rotator cuff repair or shoulder arthroplasty have been reported to be successful, including arthroscopic debridement and cyst excision or distal clavicle excision with allograft or collagen patching when joint capsule tissue is irreparable . Recurrence of the cyst after resection of the AC joint or after rotator cuff repair is rare .
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Orthopedic surgery
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Our case describes a patient who presented with a cyst of the acromioclavicular joint with initial concern for neoplastic etiology who was treated with en bloc resection of the cyst and distal clavicle. The benign nature of the cyst was confirmed by surgical pathology and histologic examination. Our case serves to emphasize the importance of performing a stepwise workup in evaluation of an atypical presentation of an acromioclavicular cyst and employing oncologic resection principles in the resection of such a cyst when etiology of the lesion is uncertain.
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Orthopedic surgery
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Acute aphasia is a very common condition that is associated with many neurological diseases such as hypoglycemia, drug abuse, and electrolyte abnormalities. However, hyperglycemia is less frequently associated with aphasia. While repetitive focal motor seizures and epilepsia partialis continua are the common presenting symptoms of nonketotic hyperglycemia 1 , isolated aphasic seizures are less frequent and very difficult to diagnose without advanced investigations 2 . We report a case of hyperglycemia-induced aphasia from non-motor seizures in the dominant cerebral hemisphere. We also review the clinical manifestations, investigations, and management of this condition from previous literature.
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Neurology
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The initial examination showed a patient with a body mass index of 17.36 kg/m 2 . All of his vital signs were normal. The initial neurological examination demonstrated a mildly confused patient. Even though he was able to follow only easy one-step verbal commands, he could simply follow body language commands without any effort. He could also remember mealtimes and attending staff correctly. Thus, delirium was less likely the main issue of his overall symptoms. The language fluency assessment showed an obviously decreased speech rate with frequent utterances and neologisms. He was unable to perform any naming, repetition, reading, and writing tests. The cranial nerve and motor examinations were all unremarkable. His initial plasma glucose was 557 mg/dL. Other laboratory data included serum osmolarity 294 mOsm/L, sodium 129.4 mmol/L, and bicarbonate 27.1 mmol/L. Other electrolyte levels, serum ketone, and arterial blood gas were within normal limits. His hemoglobin A1C was 16.5%. The results of cerebrospinal fluid (CSF) and serum polymerase chain reaction (PCR) panel for viral encephalitis and autoimmune encephalitis were all negative. Therefore, an initial diagnosis of global aphasia and type 2 diabetes mellitus (DM) was suspected.
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Neurology
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His magnetic resonance imaging (MRI) of the brain showed cortical hyperintense and subcortical hypointense T2/FLAIR lesions at left medial occipitotemporal gyrus, left occipital, and left medial temporal lobe structures. Minimal restricted diffusion with reduced apparent diffusion coefficient (ADC) was seen at left medial temporal through left medial occipital cortices. No Gadolinium enhancement was seen on the lesion . The presence of extensive vasogenic edematous lesion with minimal restricted diffusion made the possibility of arterial ischemic lesion less likely. No significant lesions in the internal carotid and vertebrobasilar systems were found in the magnetic resonance angiography (MRA) of the brain.
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Neurology
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The provisional diagnosis of isolated aphasic status epilepticus from nonketotic hyperglycemia was suggested. Saline infusion and subcutaneous insulin treatment were performed. However, subsequent EEG still showed two subtle electrographic seizures from the same area. Then, intravenous levetiracetam 2,000 mg/day was prescribed. After intensive blood glucose treatment, the patient’s symptoms gradually improved with fluent short-sentence speech and better comprehension. After treatment, his EEG had improved to intermittent theta slowing at the left posterior temporal area without an electrographic seizure.
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Neurology
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Case 1: Female, 66 years old, body mass index (BMI) of 23, with a history of hypertension and diabetes mellitus . Routine abdominal ultrasound revealed hepatic nodules. During the diagnostic workup, the patient developed ascites and an increase in the number or size of the lesions. A liver lesion biopsy was performed under interventional radiology guidance, and the histopathological analysis confirmed well-differentiated iCCA. Staging radiological examinations were conducted, including positron emission tomography-computed tomography scan (PET-CT), showed no evidence of extrahepatic disease, while magnetic resonance imaging (MRI) revealed a suspicious lymph node in the hepatogastric ligament. Neoadjuvant chemotherapy with Gemcitabine and Cisplatin was initiated. The patient completed a total of 15 cycles and achieved lesion reduction. No extrahepatic disease was found, and laparoscopy with intraoperative frozen section biopsy of the suspicious lymph node showed no evidence of malignancy. However, macroscopic cirrhotic changes were observed in the liver, despite negative serologies. Esophagogastroduodenoscopy revealed esophageal varices, and Doppler ultrasound diagnosed portal vein thrombosis ( Table 1 ). The case was referred for evaluation by the Transplant Technical Chamber and approved for living donor liver transplantation. During exploratory laparotomy, the recipient underwent a thorough inspection for extrahepatic disease. The patient received a right lobe graft donated by her son. Venous reconstruction of V5 and V8 was performed using a cadaveric iliac artery graft during the backtable procedure . Total hepatectomy was performed, along with lymphadenectomy of stations 7, 8, 9, 10, 12, 13, 16a2, 16b1, and 17. Due to portal vein thrombosis, open mechanical thrombectomy and temporary portocaval anastomosis were carried out until graft retrieval from the donor. Implantation was performed using the piggyback technique, and biliary reconstruction was accomplished through bilioenteric Roux-en-Y anastomosis. The histopathological analysis of the specimen revealed cholangiocarcinoma metastasis in 2 of 7 lymph nodes in the hepatic artery, along with lymphovascular and perineural invasion. Postoperatively, the patient presented metabolic ileus on the tenth day, which resolved spontaneously. The patient was discharged on the twentieth day without further complications. Quarterly laboratory and radiological evaluations were conducted for follow-up purposes. The patient has now completed 23 months without recurrence.
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Hepatology
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Case 2: Female, 54 years old, BMI of 23, previously treated for hepatitis C with Metavir. The patient presented a single lesion involving segments I and VIII, in close contact with the inferior vena cava, with no evidence of extrahepatic disease. A diagnosis of cholangiocarcinoma was confirmed through a percutaneous biopsy performed by interventional radiology. Initially, surgical treatment was proposed, and during the procedure, a cirrhotic liver and two satellite lesions in segments III and VIII were identified and confirmed by frozen section examination. No signs of extrahepatic disease were identified, and perihilar hepatic lymph nodes were biopsied, showing no evidence of malignancy on frozen section examination. The patient was then referred for neoadjuvant chemotherapy and immunotherapy with Gemcitabine, Cisplatin, and Durvalumab. After 6 months and 9 cycles of chemotherapy, the disease remained stable, and the patient was approved by the Transplant Technical Chamber for living donor liver transplantation. Total hepatectomy was performed using the cross-clamp technique due to the lesion’s proximity to the retrohepatic vena cava. On the back table, after perfusing the right lobe graft, venous reconstruction was performed for the dominant drainage of segment VIII using a polytetrafluoroethylene (PTFE) graft (neohepatic vena cava), and the graft reperfusion occurred uneventfully, followed by arterial and biliary anastomosis without complications. Histopathological analysis of the specimen showed clear margins, absence of lymph node metastasis, and no lymphovascular or perineural invasion. There were no postoperative complications, with a hospital stay of 11 days. A liquid biopsy performed four weeks later showed no evidence of malignancy. Radiological and laboratory evaluations after six months revealed no signs of recurrence.
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Hepatology
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Hospital Dona Estefânia, the largest pediatric hospital in Portugal, performs numerous imaging exams daily with anesthetic support. Most procedures are carried out under inhaled sedation with sevoflurane through a facial mask. However, for patients with facial dysmorphia or complex airway anomalies, the standard anesthetic approach can be risky, requiring adjustments to ensure safety and effectiveness . This case report discusses the use of the Ketodex sedation protocol for a pediatric patient with a large facial mass and subsequent predictable difficult airway, highlighting its advantages in maintaining spontaneous ventilation and minimizing airway manipulation risks.
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Anesthesiology
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