A 55-year-old male with comorbidities of diabetes mellitus, chronic obstructive pulmonary disease, and hypertension presented at our hospital with a large area of irregular, ulcerated, inflamed, circumferential marked thickening in the skin of the lower half of the left leg, with hyperpigmentation and focal areas of bleeding. In addition, he complained of pruritus. While he gave a history of eczematous dermatitis at the same site of many years, there was a significant deterioration in the past few months. Apart from the skin lesion, clinical examination also revealed left inguinal lymphadenopathy. Suspecting skin malignancy based on clinical findings, a biopsy was done which was inconclusive. 18F-FDG PET-CT was then performed to characterize the skin lesion and assess extent of the disease [Figure -]. It showed18F-FDG-avid ulceroproliferative circumferential cutaneous thickening involving the lower half of the left leg, with a length of 19.7 cm and the maximum thickness of 1.9 cm (maximum standardized uptake value [SUVmax]: 11.5) along with18F-FDG avid left inguinal lymphadenopathy, measuring 2.7 cm × 2.6 cm (SUVmax: 5.2). No other metastatic lesion was suspected in the rest of the body. The patient was then planned for surgical excision of the skin lesion with left inguinal nodal dissection, after on table frozen section. The patient refused surgery at that time and lost to follow-up. He presented eight months later with the same complaint, but increased oozing from the skin lesion. 18F-FDG PET-CT was again performed for restaging, before planning the surgery [Figure -]. The second PET-CT showed similar findings as before, with no significant change in length (18.9 cm) and some increase in the thickness of cutaneous lesion (2.1 cm) and no significant change in nodal size (2.6 cm × 2.5 cm); however, interval reduction of18F-FDG uptake was seen at both sites (SUVmax: 9.1 and 4.6, respectively). Since there was a reduction of18F-FDG uptake and relatively stable size without any anticancer treatment over a relatively long follow-up period, suspicion of some benign skin pathology was raised on PET-CT, and rebiopsy was advised. Repeat biopsy from the skin lesion showed hyperkeratosis, focal parakeratosis, acanthosis, papillomatosis consisting of horn cysts, and exocytosis in the epidermis, with dermis showing moderate perivascular and periadnexal chronic inflammatory infiltrate and edema, suggesting irritated seborrhoeic keratosis. There was no evidence of dysplasia or malignancy. Fine-needle aspiration cytology from the left inguinal node also showed inflammatory changes. The patient was then managed conservatively, with symptomatic improvement. He is scheduled for ablative therapy for the skin lesion.


A 50-year-old female patient with no associated diseases, who was a non-smoker, underwent lipoabdominoplasty. In the immediate postoperative period, she wore a low-compression mesh (8 mmHg) and an anatomical flexible polyurethane abdominal splint (R-Slim). Infrared thermography was performed 24 hours after the operation and on days 5, 25, and 27 postoperatively.


A 46-year-old female patient with no associated chronic diseases, who was a non-smoker, underwent lipoabdominoplasty and mastopexy with a prosthesis. She wore a low-compression (8 mmHg) mesh in the immediate postoperative period. Infrared thermography was performed 24 hours after the operation and on days 5, 25, and 27 postoperatively.


A 35-year-old African American female with medical history of HIV, off antiretroviral therapy (ART) for over a year, chronic kidney disease Stage 3b, membranoproliferative glomerulonephritis secondary to HIVICK that was recently diagnosed by biopsy, and pulmonary hypertension presented to the emergency department complaining of burning sensation in her feet, bilateral swelling of her legs along with difficulty walking and shortness of breath with exertion. She denied any cough, chest pain, palpitations, fever, or sick contacts. Vital signs on admission showed a temperature of 36°C, heart rate of 95/min, blood pressure of 150/100 mmHg, respiratory rate of 18/min, and oxygen saturation of 100% on 2 L/min of oxygen through nasal cannula. Physical examination was significant for engorged neck veins, elevated jugular venous pressure, distant heart sounds, decreased air entry bilaterally with basal crackles, and 3+ lower extremity pitting edema up to the knees. Laboratory workup showed bicarbonate level of 17 mmol/L (20-31 mmol/L), blood urea nitrogen of 26 mg/dL (6–24 mg/dL), serum creatinine 1.7 mg/dL (0.5–1 mg/dL), and estimated glomerular filtration rate of 34 mL/minute/1.73m 2. Cardiac markers such as brain natriuretic peptide was 507 pg/mL (0–100 pg/mL) and initial troponin I 0.485 ng/mL (0–0.45 ng/mL). Inflammatory markers such as D-dimer were elevated, 2300 ng/mL (0–500 ng/mL), but C-reactive protein and procalcitonin were within normal limits. CD4 count was 173/uL (359–1519/uL) with viral load of 30,780 copies/mL. She had negative Hepatitis B surface antigen, and negative Hepatitis C antibody; her blood cultures were negative. Urine analysis showed nephrotic range proteinuria (+3). Chest X-ray (CXR) showed small bilateral pleural effusions with enlarged cardiac silhouette with “3 bottle” configuration suggestive of pericardial effusion []. Echocardiogram showed significant pericardial effusion with signs of tamponade [], right ventricular systolic pressure was elevated at 64 mmHg (normal range: 15–25 mmHg), for which she was transferred to cardiac critical care unit for close monitoring. The next day, she underwent an emergent pericardiocentesis with pericardial window, 800 cc of serosanguinous fluid was drained from the pericardium. A chest tube was also inserted to drain the right pleural effusion with an output of 1000 cc. The pericardial fluid analysis revealed transudative nature, cultures were negative and acid-fast bacillus test was negative too. Cytology showed no evidence of malignancy and the pericardial tissue pathology showed mild focal inflammation with no evidence of malignancy. Afterward, the patient was monitored in the CCU for a few days, there were no complications of the procedure, repeat CXR showed resolution of the pericardial effusion and improvement in the left pleural effusion []. The patient was discharged, with outpatient follow-up with cardiology, nephrology, and infectious disease departments.


During the routine dissection of a male cadaver aged 75-year-old at death, a venous sinus () was identified traveling from the superior orbital fissure anteriorly, over the floor of the middle cranial fossa, over the petrous part of the temporal bone, to drain into the transverse sinus. The variant was found on left and right sides () but the right sided sinus was damaged before it could be photographed. Anteriorly, the sinus communicated with the superior ophthalmic vein, which drained primarily into the cavernous sinus. Along the floor of the middle cranial fossa, the venous sinuses communicated with the middle meningeal veins (). The superior petrosal sinus traveled medial to the sinus and did not communicate with it. The variant venous sinus was approximately 8 mm in width at its anterior and posterior extremes but was dilated to 1.2 cm as it crossed over the petrous part of the temporal bone. The sinuses were roughly 7.2 cm long. No additional anatomical variations or intracranial pathology were noted in this specimen.


A 38-year-old male was initially seen by the ophthalmology service due to blunt trauma to the right eye secondary to assault with a metal pipe. On initial consultation in the hospital, he was diagnosed with a right-sided closed orbital wall fracture, traumatic iritis with microhyphema, and a choroidal rupture in the right eye. Vision at that time was hand motion, and intraocular pressure (IOP) was 44 mmHg through rebound tonometry. After initial treatment with topical steroids, his visual acuity improved from hand motion to 20/60 over the course of 2 months. During his recovery, the patient was noted to have persistent hypotony and macular folds []. No cyclodialysis cleft was initially seen, although he was persistently dilated on cycloplegia for the iritis during initial gonioscopy. In addition, his IOP was noted to be consistently lower than the left eye, with at least a 5 mmHg difference each visit, and ranged between 6 and 11 mmHg. A month after his initial injury, choroidal folds were noted on examination and on optical coherence tomography. Although his IOP at this time was 9 mmHg, the IOP in his left eye was 20 mmHg.
Three months after his initial injury, he was referred to the retina service for the evaluation. Best-corrected visual acuity (BCVA) was 20/60 in the right eye, IOP was 11 mmHg in the right eye, and 20 mmHg in the left eye. Examination of the anterior segment revealed a shallow anterior chamber, and undilated gonioscopy revealed a 5-clock-hours cyclodialysis cleft located temporally. The patient was then referred to the glaucoma service for cyclodialysis cleft closure. Initial surgical repair was performed by direct cyclopexy through scleral flaps with 10–0 prolene suture. Afterward, a small area (<1 clock h) of open cleft was still noted superotemporally, which did not close after application of argon laser. A repeat direct cyclopexy was performed a month after initial surgery with 9–0 prolene suture, but was unsuccessful in relieving his hypotony.
Given his refractory course, he was referred to another glaucoma specialist for evaluation and treatment. Ultrasound biomicroscopy (UBM) examination revealed shallow choroidal serous detachments with an atrophic, detached ciliary body processes corresponding to the location of the cyclodialysis cleft. Gonioscopic examination showed an area concerning for residual cleft opening between 7 and 8 o'clock, but closure of the cleft was confirmed with pressure on the globe during UBM []. His hypotony was thought to be secondary to infarcted ciliary body processes secondary to detachment. Due to the cause of hypotony, the glaucoma specialist suggested phacoemulsification with capsular tension ring (CTR) insertion with the IOL, and pars plana vitrectomy with gas tamponade to bolster and reattach the ciliary body as a more heroic measure.
A month afterward (over 3 months since the initial injury), a combined phacovitrectomy with IOL, CTR implantation, and gas tamponade with 22% sulfur hexafluoride (SF6) was then performed. During phacoemulsification, zonular instability was noted in areas associated with the cyclodialysis cleft. A MORCHER® Type 14C CTR (FCI Ophthalmics, Pembroke, MA, USA) was inserted into the capsular bag to provide capsular support, and an +22.50D SN60WF acrylic monofocal IOL (Alcon, Fort Worth, TX, USA) was placed in the capsular bag without complication. The diopter was chosen based on the calculations and axial length of the normal contralateral eye. A complete vitrectomy was performed, with induction of a posterior vitreous detachment and vitreous skirt shaving. After air-fluid exchange, the air was exchanged with 22% SF6 in the vitreous cavity. He was then instructed to remain supine for a week. Postoperatively, the patient had short-term ocular hypertension above 30 mmHg that was well controlled with topical therapy and oral acetazolamide. A little over 1 month after the surgery, the hypotony resolved in the right eye, with normalization of the anterior chamber and ocular hypertension at 28 mmHg on topical steroids. His topical steroids were tapered off over the next 2 weeks, and his pressure decreased to 14 mmHg without any topical aqueous suppressants. Three months after the surgery, his BCVA improved to 20/25 (requiring +0.50 D correction) and his IOP was 14 mmHg without any medications, with some resolution of his choroidal folds. His cyclodialysis cleft was noted to be closed on gonioscopy at this visit. A year after his last surgery, BCVA remained 20/25 without any complications, with a measured IOP of 18 mmHg off all drops. No lens decentration or dislocation was noted at present. His choroidal folds were noted to have improved significantly, though not completely resolved [].


An 8-month-old female baby was delivered through cesarean section after full-term pregnancy (37 weeks) one of monozygotic twins; her mother had a bleeding during second trimester of pregnancy; her hemoglobin dropped to 7 g/dL and was managed by blood transfusion of two units. The baby's weight at the time of delivery was 2.5 kg, with APGAR score of 7 and 9 in 1 and 5 min, respectively. The baby was accompanied by her mother to the primary care clinic with multiple hyperpigmented lesions that had been present since birth and were increasing in number (>9). The baby was the result of in vitro fertilization. Regarding her developmental history, gross motor function: she could crawl and sit without support. Fine motor: she could transfer things from one hand to another. Language: she could say some words like “mama,” and understood such words as “No” said to her. There were no developmental delays compared to other patients of that age group. The patient was breastfed only for the first 75 days after delivery as the mother's breast milk secretion was interrupted thereafter. There was no history of fever, itching, redness, conjunctivitis, dry eye, bowel changes, fatigue, weight loss, or neurological deficits. The patient is a twin, and vaccinations were up to date.
Dermatological examination revealed several hyperpigmented macules on the skin with a diameter of >1.5 cm and a number of 10–20 on the chest, abdomen [], and back []. There was no axillary or inguinal freckling, and mucous membranes were unaffected. Ophthalmological examination found that the lacrimal system, conjunctiva, cornea, and anterior chamber were within normal limits. Bilateral Lisch nodules were not seen in the iris of either eye. Fundus examination revealed no abnormality in the right and left eye. No retinal breaks or retinal detachment was seen bilaterally. An annual follow-up was suggested for that.


A 3-day-old healthy baby girl with a birth weight of 2.8 kg and a length of 48 cm, delivered full term through vaginal delivery from a multigravida mother, presented with congenital absence of the skin on the scalp, back, buttocks, and upper limbs with an atrophic scar on the chest and abdomen. The mother was currently 40 years old and had four children from a previous pregnancy. During antenatal care (ANC), the mother had no history of illness, trauma, or taking medicine other than vitamins prescribed by the doctor. Ultrasonography was performed at the third trimester and showed a single live fetus with no obvious congenital malformations. Soon after delivery, the mother had vaginal bleeding due to retained placenta. The bleeding did not stop despite subsequent curettage, and a hysterectomy was performed. Histopathological examination of the uterus showed placenta accreta with no signs of infarction. All the previous pregnancies were full-term, with no complications, and all her children were delivered vaginally. A history of miscarriages was denied. The first child was a male, died at the age of 2 months due to diarrhea, the second is a female, currently 16 years old, the third is a boy – 14 years old, and the fourth is a male – 7 years old. There was no family history with a similar skin condition. Upon physical examination, the lesions were well demarcated, symmetrical, and stellate, covered with a thin, gelatinous, shiny membrane. The lesions on the trunk were distributed in an H-shaped pattern measuring 20×7 cm, while lesion on the scalp was 6×4 cm. Atrophic scar, 14×5 cm in size, was found in the chest and abdomen (). There were no embryological malformations such as meningomyeloceles, spinal dysgraphia, cranial stenosis, congenital midline porencephaly, leptomeningeal angiomatosis, ectopia of the ear, omphalocele and gastroschisis, blistering, nail deformity, or malformation syndrome. Laboratory examination revealed hypoalbuminemia (2.26 g/dL) and elevated C-reactive protein (6.73 mg/dL), while other parameters were within the normal limit. Head X-ray showed irregularity of the parietal lobe in the vertex region, while radiological examination of the trunk showed no abnormalities. Magnetic resonance imaging (MRI) with contrast in the head revealed no subcutaneous defect. The patient was consulted to otolaryngologist, ophthalmologist, neuropediatrician, and cardiologist; consultation results indicated no abnormalities other than dermatological abnormality. Initially, lesions were treated with Sorbact® gel dressing-BSN Medical for autolytic debridement, and then covered with sterile gauze until the gelatinous-shiny-membrane disappeared. Following that, we used hydrocolloid wound dressing (WD) until complete epithelialization was achieved (). Prophylactic antibiotic was given with intravenous injection of ampicillin sulbactam and gentamicin. We also gave 20% albumin due to hypoalbuminemia until the normal level was reached. Bacterial culture from wound base was positive for Klebsiella pneumoniae with resistance to ampicillin and gentamicin. Therefore, antibiotic treatment was modified to intravenous injection of meropenem. After 26 days, the patient was discharged from neonatal intensive care unit (NICU) when more than 70% of the lesion showed epithelialization. Following daily local treatment, full epithelialization was achieved at day 72, leaving a smooth, hairless, papery scar (). Until the age of five months, no medical problems were apparent, and the infant showed completely normal motor skills development.


This 38-year-old woman had dysphonia for 1 year when she first attended our service. At onset, there had been a change in the quality of her singing voice over the course of a week. Her voice was dysphonic but had preserved articulation. No infective symptoms were recalled. She had also been aware of intermittent facial twitching, mainly around her eyes during times of stress, or after drinking coffee. Soon after the onset of her dysphonia, she was examined by an ENT surgeon and a neurologist. Fibreoptic naso-endoscopy revealed an abducted left vocal cord with only flickers of movement. The rest of the neurological examination was normal. MRI of her brain and CT of the chest and neck looking for a structural cause were unremarkable.
There was a strong family history of MND/ALS with her father, paternal aunt and uncle all affected. Genetic testing in her father had demonstrated an SOD1 mutation (Val149Gly). Our patient had elected not to proceed with predictive gene testing.
Over the first 12 months, her dysphonia gradually worsened. She had difficulty with coughing and developed dysphagia to some solids, such as bread, with a feeling of food stuck in the pharynx. There were a few occasions of nasal regurgitation when trying to swallow. Although she described intermittent exertional breathlessness, spirometry showed a forced vital capacity (FVC) of 125% predicted. At this point, she sought genetic counselling and proceeded to genetic testing. Before the test results were known, the geneticist referred her to the MND service for assessment, due to concern over her symptoms.
Neurological examination at that time revealed severe dysphonia, with a nasal, breathy voice but no dysarthria. The cough was bovine and there was left palatal weakness. Occasional fasciculations were present in the periocular muscles. The rest of the neurological examination was normal. Repeat naso-endoscopy confirmed a failure of the soft palate to close completely on the left-hand side. The left vocal cord was now paralysed. A repeated MRI of the brain and cervical region showed no abnormalities. Over the subsequent month, the patient appeared to have an accelerated deterioration in her condition with increased dysphonia, more widespread facial fasciculations and the onset of fasciculations in her shoulder girdle. Examination confirmed this along with new weakness in the intrinsic muscles of her left hand and left-finger extensors. Evidence of upper motor neuron involvement was also present with brisk reflexes in the left arm. Electromyography demonstrated sparse fibrillation potentials and minor neurogenic changes in the arms and simple fasciculation potentials in the tongue. Nerve conduction was normal. Repeated respiratory function showed that her FVC was now 84% predicted, and maximal respiratory pressures were reduced. Genetic testing results confirmed the presence of an SOD1 mutation (Val149Gly). Serial neurological examination a month later showed further deterioration with mild wasting of the tongue along with fasciculations, increased wasting in the left hand and pathologically brisk reflexes of the left upper and lower limbs. Following discussion, the patient elected to explore non-invasive ventilation, percutaneous endoscopic gastrostomy and injection thyroplasty to assist with symptom management.


A 17-year-old girl presented with abnormal posturing of the right upper and lower limbs since the age of 9 years, followed by involvement of left upper and lower limbs at the age of 11 years, difficulty in speaking at 13 years and abnormal neck posturing noted at 15 years of age. At the time of presentation to us, she had generalized dystonia involving the lower face, tongue, neck, trunk, and all four limbs. A bulbous nose tip was noted [See ]. She was anarthric and wheelchair bound. She had normal cognition and no evidence of parkinsonism, spasticity, incoordination, extraocular movement, or other neurologic deficits. Birth and developmental history were uneventful. Her elder sibling had early onset generalized dystonia [See for pedigree details].
Biochemical investigations including workup for Wilson's disease were within normal limits. Cranial MRI revealed symmetrical T2 hyperintensities in bilateral posterior putamen without any evidence of mineral deposition. [Figure -]. Written informed consent was obtained from the patient for publication. Clinical Exome Sequencing revealed a novel heterozygous missense variant, c.4675G > T (p.D1559Y) in exon 20 of the KMT2B gene []. To confirm it further, we designed primers covering exon 20 of KMT2B gene and performed Sanger's sequencing of the proband along with her parents. Parental screening revealed the same variant in heterozygous state in her asymptomatic mother []. The variant, p.D1559Y was classified based on the joint guidelines of ACMG (American College of Medical Genetics and Genomics) and AMP (Association of Molecular Pathology) and reported as VUS (Variant of Uncertain Significance). Multiple lines of computational evidence suggested a deleterious effect on the gene or gene product. This variant was also absent from, 1029 genomes from India (IndiGenomes), SAGE (South Asian Genomes and Exomes), and other global databases including ExAC, ESP6500, GME, 1000G but observed in heterozygous form in 1 South Asian male out of 15301 samples considered for gnomAD exome with minor allelic frequency 0.000004014 (gnomAD exome all) and 0.00003268 (gnomAD exome SAS).
She had previously received levodopa upto 300 mg/day without any benefit. She was intolerant to anticholinergic drugs and was presently on tetrabenazine 75 mg/day and clonazepam 0.25 mg/day with persistent significant disability. The Burk-Fahn-Marsden Dystonia Rating Scale motor score (BFMDRS-M) was 94 (range 0-120) and BFM-Disability (BFMDRS-D) score was 24 (range 0-30). Considering the severe disability and lack of response to medical management, she was offered GPi-DBS. She underwent stereotactic guided bilateral GPi-DBS with microelectrode recording and clinical monitoring for adverse effects under general anesthesia (Activa RCÒ, Medtronic, Minnesota). Immediately after surgery, there was mild improvement in the neck and limb dystonia. At 6 months follow up, after optimization of stimulation parameters, the BFMDRS-M score was 57.5 and the BFMDRS-D was 22. [See ] She had regained ambulation with support. [See ] At last follow-up, stimulation parameters for left GPi were 3(-) C (+), 1.5 V/60 μs/150 Hz and for right GPi were 11(-) C (+), 1.7V/90 μs/150 Hz.


A 68-year-old man diagnosed with carcinoma of pyriform fossa, with no comorbidities was admitted to our intensive care unit (ICU) with hypoxemic respiratory failure and septic shock. The trachea was intubated and fluid resuscitation and vasopressor support were started. A 7 Fr sized triple lumen central venous catheter (CVC) was inserted in the right IJV under ultrasound guidance and in the first attempt successfully without any complication. Over a few hours, the patient was observed to have life-threatening complications of acute renal failure (anuria, metabolic acidosis with HCO3 of 12 mEq/L, and hyperkalemia with K + of 6.8 mEq/L). Therefore, urgent hemodialysis was planned for the patient, and consent for the same was taken from the patient's relatives. The left IJV was chosen for dialysis catheter insertion for two reasons; first, the right IJV was already catheterized with CVC, and second, the femoral vein cannulation is associated with higher risks of catheter-related infection, thrombosis, and occlusion as compared to IJV cannulation.
The patient was positioned with the head slightly lower than the rest of the body and the left side of the neck was cleaned with 2% w/v chlorhexidine gluconate and draped. We planned to follow the Seldinger technique under ultrasound guidance. Local anesthesia was given at the puncture site with 4 ml of 2% lignocaine. After locating the IJV using ultrasound (linear probe, 5 MHz frequency, Sonosite Edge II), the vein was punctured at 45° angle with the introducer needle and free aspiration of blood was confirmed. Following a successful puncture, the guidewire was threaded through the needle. After 5 cm length of guidewire was threaded, resistance was felt. Further attempt to thread the guidewire was stopped and a guidewire was removed. The syringe was connected to needle hub and free blood aspiration was done to confirm the presence of the needle in vein. On reattempting the guidewire insertion through the needle, we felt the resistance after 5 cm length of guidewire was inserted. We confirmed bilateral normal breath sounds, a normal respiratory rate, and oxygen saturation of 98% with no evidence of subcutaneous emphysema or hematoma, venous congestion, or limb ischemia. At this point of time, the senior ICU registrar was called for help.
Without removing guidewire, needle and guidewire were scanned to check the position of a guidewire in relation to the vein. Guidewire with the needle was found to be inside the vein; however, a loop of guidewire was visualized which was abutting the posterior wall of the vein in out-of-plane (axial) view, creating a dual-point echogenicity []. We retracted back the introducer needle a bit making sure that the guidewire was still in its place. Under real-time ultrasound guidance, we slowly pulled out the guidewire till the loop (dual-point echogenicity) disappeared; making sure that the guidewire was visible in the vessel lumen all the time. Again, the guidewire was advanced caudally in the vein, which could be threaded further without any resistance. Following dilatation, the dialysis catheter was railroaded over the guidewire, and a guidewire was removed. Correct intraluminal placement of dialysis catheter in the left IJV was conformed using ultrasonography (USG) []. Blood was aspirated in both ports and free flow was confirmed. A bedside chest X-ray was performed to ensure the correct placement of the hemodialysis catheter.


A four-year-old female presented to our pediatric rheumatology clinic with the chief symptom of periodic fevers and abdominal pain.
The patient’s fevers began 4 months prior to presentation following a documented streptococcal pharyngeal infection, with a maximum temperature of 104°F. They occurred every 1–2 weeks and would then last a duration of 4–6 hours. Fevers were followed by severe abdominal pain that prevented eating. She did not have associated emesis, diarrhea, or constipation.
The past medical history of the patient included allergic rhinitis, characterized by rhinorrhea and congestion, which was treated with nasal spray. Otherwise, she was reported to have normal growth and development. There was no known family history of autoimmunity, including inflammatory bowel disease, systemic lupus erythematosus, vasculitis, psoriasis, or Raynaud’s syndrome. Social history was noncontributory. There were no known relevant environmental exposures.
Review of systems was notable for lack of joint pain or swelling, digital clubbing, myalgia, limp, rash, fatigue, or headaches. The patient’s parents denied that she had experienced respiratory symptoms, including shortness of breath, dyspnea on exertion, cough, or snoring. There was no history of recurrent respiratory infections.
The girl was a well-appearing child without dysmorphic features. Vital signs were normal, though pulse oximetry was not obtained initially. There were no oral lesions, the oropharynx was clear, and tonsils were without exudate. Findings of ophthalmic examination were normal. Cardiac examination did not reveal murmur, rub, gallop, extra heart sounds, or arrhythmia. Lung examination demonstrated normal and symmetric air entry bilaterally. Her abdomen was soft, nontender to palpation, and without hepatosplenomegaly. Musculoskeletal examination was notable for redness overlying the distal interphalangeal joints and bilateral finger and toe clubbing (Figure ). There was no evidence of synovitis or muscle tenderness or weakness. The patient had a normal gait without evidence of limp.
Results of initial laboratory tests were notable for an elevated level of alkaline phosphatase at 1,059 units/liter, but otherwise normal for transaminases, complete metabolic panel (albumin level of 6.5 gm/dl and bilirubin level 0.6 mg/dl), complete blood cell count, sedimentation rate, S100A8/S100A9, S100A12, complement levels, immunoglobulin levels, and C-reactive protein level (obtained between fever episodes). Antinuclear antibodies were negative.
A chest radiograph demonstrated nonspecific peribronchial thickening.


A 39-year-old man with a history of drug abuse attempted intentional self-harm with a pneumatic nail gun by shooting himself multiple times in his left anterior chest and the right side of his head. After the attempt, he complained of a headache and chest pain, and he reached out to his mother for help. When his mother took him to the emergency department (ED), he was conscious (Glasgow coma scale score: E4V5M6) with a normal respiratory rate (RR) of 18 breaths/minute, tachycardia (155 bpm), mild hypotension (blood pressure: 94/63 mmHg), and peripheral arterial oxygen desaturation (arterial oxyhemoglobin saturation [SaO2]: 92%). One visible nail head was found within the right temporal area (), and three were found in the left chest () approximately 2 cm above the left nipple. The trauma protocol was initiated immediately to provide immediate treatment and resuscitation. Focused assessment with sonography for trauma (FAST) examination revealed pericardial effusion. Whole-body computed tomography (CT) revealed three metallic nails penetrating the left lower anterior chest wall through the lung to the heart, with significant hemopericardium, mild pneumothorax, and moderate hemothorax (). Brain CT showed one metallic nail that had punctured the right temporal bone without intracranial hemorrhage (ICH) (). After consulting with a cardiac surgeon, chest surgeon, and neurosurgeon, emergency surgery was arranged.
Via a median sternotomy, the cardiac surgeon opened the pericardium to identify two rupture sites on the left ventricle (LV) adjacent to the left anterior descending (LAD) artery bifurcation. Each rupture wound was repaired using pledgeted 4-0 Prolene sutures (Ethicon Inc, Somerville, NJ, USA) (). During a sternotomy approach to remove three 5-cm-long steel nails, through-and-through penetration of the left upper lung lobe was noted, and wedge resection was performed by the chest surgeon. Copious normal saline irrigation was performed after the heart and lung repair. Postoperatively, the patient was admitted to the surgical intensive care unit for further resuscitation.
Eight hours after being admitted to the ICU, the patient’s hemodynamic status was stable under constant sedation, without the need to administer vasopressors or anti-arrhythmic agents. After initiating valproic acid to address focal seizures, we performed a craniotomy around the nail in the right temporal bone (). The nail puncture hole in the brain was contiguous with two branches of the superficial cerebral vein (); however, no vessels were injured by the nail. There was mild oozing from the brain after removing the nail, without a significant hematoma. Postoperatively, levetiracetam was added to the valproic acid to address recurrent focal seizures. The patient was extubated on postoperative day 1 and was transferred to the general ward on postoperative day 3, fully conscious. In the general ward, the patient’s vital signs and consciousness level remained stable. Limb weakness, dysphagia, dysarthria, or arrhythmia were not observed. We consulted the psychiatric department to obtain drug abuse evaluation for the patient, and consulted social workers for discharge planning. Fourteen days after transfer to the general ward, the patient was discharged. At the 2-month follow-up after discharge, the patient had achieved 100% of his baseline performance, without cognitive, motor, or respiratory symptoms.
The study protocol was approved by the Institutional Review Board of Chang Gung Medical Foundation (approval number: 20200947B0). We have obtained consent from the patient to publish this report. The reporting of this study conforms to the CARE guidelines.


A 50-year-old female with metastatic duodenal cancer began experiencing worsening control of her diabetes after exposure to an immunotherapy combination Phase 1 clinical trial. Her prior medical history included a new-onset Type 2 diabetes 6 months before her cancer diagnosis. She underwent a Whipple procedure after which she was initiated on lantus insulin that continued for 4 months and then transitioned to oral therapy with metformin 500 milligrams twice daily thereafter. She was followed by her local endocrinologist initially; however, due to worsening glucose control, a referral was placed to the endocrinology department for assistance with diabetes management. Her hemoglobin A1c (HbA1c) was 6.8% before initiation with pembrolizumab, which increased to 8.5% after five cycles and 9.2% after nine cycles of treatment. She began experiencing extreme fasting blood sugars in the 300's and occasional hypoglycemic episodes in the low 50's at night, along with tachycardia and diaphoresis. Polyuria and polydipsia were also present during the episodes with hyperglycemia. During the time of the consult, the endocrinology team found that her HbA1c was 10.4% with a connecting peptide (C-peptide) of less than 0.02; however, her islet cell antibodies were nonexistent in the blood work. These laboratory data along with the pattern of her presentation were highly suggestive that she had developed IDDM secondary to immunotherapy. She was initiated on tandem insulin pump, Tresiba and metformin 1000 milligrams twice daily in addition to using a premeal insulin sliding scale. She was taken off Phase 1 clinical trial after 15 cycles secondary to disease progression and was transitioned to standard of care treatment.


In February 2021, a >60-year-old male journalist was hospitalised for a massive right pneumothorax. He was a social drinker with a physically active lifestyle. At a young age he had been a heavy smoker (from 25 to 50 years of age, more than 20 cigarettes a day, 35 packs/year before quitting smoking) and had subsequently developed emphysematous chronic bronchitis. Starting from the early 1990s, he had worked as a war correspondent in the Balkans, Iraq and Afghanistan for about 10 years. Since 2003, he had been working in an urban office. Family history was positive for cancers which affected both parents (lung cancer) and two sisters (breast cancer).
When questioned about his war experience, he stated that during each mission he had lived for many months among the rubble of bombed-out buildings, constructed mainly in the 1950s and 1960s with the extensive use of asbestos. The destruction of entire urban and industrial areas had created mountains of rubble where everything was covered in dust, and it was impossible to avoid breathing it. Referring to himself and his colleagues he is reported as saying, ‘At times we felt like we were chewing that air, it was so thick and slimy’.
In 2017 he developed a mucoepidermoid carcinoma (MEC) of the soft palate and underwent surgery (partial maxillectomy and palate reconstruction). The diagnosis was confirmed by histochemical methods (diffuse p63, CK19 and CK14 phenotype, focally positive for Cam 5.2).
During the recent hospitalisation, after pleural drainage, persistent air loss and incomplete pulmonary re-expansion, a thoracoscopy and biopsies were performed. A multifocal malignant pleural mesothelioma (MM) with microfoci of initial infiltration was diagnosed. The patient underwent surgery (right apicoectomy for bullous dystrophy and chemical pleurodesis) and chemotherapeutic treatment (cisplatin and pemetrexed).


A 64-year-old postmenopausal woman (gravida 2, para 2) presented at the department of gynecology with a short history of nausea and abdominal distention. The patient had no relevant history of urogynecological malignancies and gynecological diseases. The women had no inheritable conditions and no family cancer history in first-degree relatives. The patient had no history of tobacco use or alcohol or drug consumption. She had had arterial hypertension for 6 years. Gynecological examination revealed a palpable tender mass located near the right ovary. Abdominal and vaginal ultrasonography showed a large amount of free fluid in the abdomen with hyperechoic echogenicity and a right ovarian tumor with mixed echogenicity (). A laboratory examination was normal except for anemia with 10.9 g/dL hemoglobin. The laboratory examination of tumor markers revealed elevated tumor markers – carcino-embryonic antigen (CEA) (23.4 ng/mL), carbohydrate antigen 19.9 (CA 19.9) (155.1 IU/ml) and carbohydrate antigen (CA-125) (39.4 IU/ml). Computed tomography demonstrated the presence of a heterogeneous, hypodense mass, without contrast enhancement, located on the right side of the pelvis, near the right ovary. The CT also showed central calcifications and fatty components of the tumor. Enlarged right pelvic lymph nodes and a large amount of free fluid in the abdominal cavity were also observed (). The patient underwent exploratory midline laparotomy, which revealed a large amount of yellow gelatinous mucinous ascites – approximately 1.5 l. A tumor (6 x 7 cm in diameter), arising from the appendix and located in the pouch of Douglas near the right ovary, was noted. Macroscopically the tumor was gross and fleshy without involvement of the base of the appendix. The right pelvic lymph nodes were enlarged. There were no implants on the parietal and visceral peritoneum. Evaluation for distant metastases was negative. Ascites fluid was obtained (30 ml) for analysis. Appendectomy, total laparoscopic hysterectomy with bilateral salpingo-oophorectomy (TLHBSO) and bilateral pelvic lymph node dissection were performed. Histopathology examination revealed poorly differentiated mucinous appendiceal adenocarcinoma (MAA), comprising up to 50% signet ring cells []. The tumor perforated the appendix and had grown through its visceral peritoneum (). Genital organs and lymph nodes were normal. The patient was diagnosed with surgical stage IV A according to the American Joint Committee on Cancer staging classification and pT4aN0M1aG3, according to the TNM classification (acronym: tumor, node, metastasis) []. Ascites fluid analysis showed mucin without identifiable neoplastic epithelial cells. PMP was classified as acellular PMP. Intraperitoneal lavage with 5% dextran and water was performed. We did not leave any drains in the abdominal cavity as we used enhanced recovery after surgery (ERAS) protocols []. The patient was discharged on the 10th postoperative day in a good health condition. Four months after the operations, the patient is without complains. The follow-up 3 months after the operation (gynecological examination, abdominal, vaginal ultrasonography and tumor marker levels (CAE, CA-19-9, CA-125) did not show any evidence of recurrence.


A 65-year-old woman, an ex-smoker with a Brinkman index of 800, with a past history of breast cancer, urothelial cancer, and ischemic heart disease presented cough for several months. Plain chest X-P showed a mass in the right lung field and enlargement of the right lung hilum (Fig. ). Computed tomography (CT) showed well-defined oval masses in the lung with apparent hilar lymph node enlargement. Laboratory testing showed no significant tumor marker elevations. Pathological examination of the transbronchial lung biopsy specimen showed large oval atypical cells with mucin formation in some cells. Immunostaining showed that the atypical cells (Fig. ) were TTF-1, napsin A, CK5/6, CD56, synaptophysin, and chromogranin negative, Alucian blue positive, p40 and p63 focally positive, and PD-L1 weakly, that is,1%, positive. Molecular testing showed that no molecular mutations were observed in epidermal growth factor receptor exons 18–21. Positron emission tomography (PET) showed maximum standard uptake values of 8.0 and 14.1 in the 2 tumors located in the right lower lobe and those of 6.3 and 5.3 in the hilar and mediastinal lymph nodes, respectively, but no significant accumulation in the bone (Fig. ). The patient with digital clubbing and cortical and periosteal thickening of the tibias complained of polyarthralgia after the onset of cough, leading to the diagnosis of pulmonary hypertrophic osteoarthropathy. The presence of 2 lung cancers in the right lower lobe suggested the T category in this case as at least T2, that is, more than 3 cm in size, or T3 with possible pulmonary metastasis in the same lobe. In addition, bulky presumed hilar lymph node metastasis could lead to inevitable pneumonectomy when primary surgery was applied to the patient. We, therefore, treated the patient with primary chemoimmunotherapy using pembrolizumab (400 mg q3w), carboplatin (AUC 5 mg/mL/min q3w), and pemetrexed (500 mg/m2 q3w). The patient showed prompt symptom relief, that is, disappearance of the cough and polyarthralgia with manageable side effects. Due to the quasi-complete remission of the lung cancers and the marked shrinkage of the presumed lymph node metastases (Fig. ) with 3 courses of the chemoimmunotherapy, the patient underwent lower lobe lobectomy and hilar and mediastinal lymph node dissection. Pathological examination showed no viable cancer remnants in the lung tumors and lymph nodes dissected (Fig. ). The postoperative course was uneventful and the patient was discharged on the 9th day after operation.


A 35-year-old female patient was brought to emergency with penetrating arrow injury in the dorsal aspect of the thorax and on the right third intercostal space (ICS), inflicted about 15 h ago []. There was an altercation that resulted in her being shot by a bow and arrow.
The patient was conscious and oriented, but irritable. Her heart rate was regular (100/min) and blood pressure (BP) was 90/60 mmHg. Oxygen saturation (SpO2) was recorded 95% with room air. She was dyspneic with a respiratory rate (RR) of 33/min. There was no history of any comorbidities. Hemothorax was detected on a computed tomography scan. A chest X-ray confirmed the position of the arrow tip penetrating the middle lobe of the right lung and impinging over the pericardium [].
There was decreased chest expansion to the right side. Tenderness along with decreased vocal fremitus and decreased vocal resonance were detected. There was dullness and decreased air entry over the right hemithorax. Heart sounds S1 and S2 were normal with no murmur. Glasgow Coma Scale was 15/15.
On airway examination, mouth opening was detected to be of three fingers breadth, and Mallampati Grade was II. The thyromental distance was within normal limits. The temporomandibular joint was normal. There were no loose teeth or removable dentures.
Before transferring to the operation theater (OT), a chest tube was inserted in the 5th, right ICS. In OT, IV access was established by two 16G IV cannulas. Routine monitors of electrocardiography, noninvasive BP (NIBP), and pulse oximetry (SpO2) were connected. A decision to intubate the patient in the semi-left lateral position was taken as the arrow had lodged in the right lung.
The patient was premedicated with injection glycopyrrolate 0.2 mg, injection midazolam 1.5 mg, injection nalbuphine 10 mg, injection pantoprazole 40 mg, and injection ondansetron 4 mg and preoxygenated at 6 L/min for 3 min. Induction of general anesthesia was achieved with injection etomidate 16 mg and injection scholine 75 mg.
The patient was intubated with a 35 fr of the double-lumen tube (DLT) on the left side in a semi-left lateral position. The position was confirmed by end-tidal CO2, auscultation, and fibroscope. It was subsequently fixed at a 28 cm mark at the left angle of the mouth.
Air and oxygen were maintained at a rate of 50:50. Isoflurane was given intermittently at the rate of 1 Mac. injection vecuronium 5 mg IV was given at loading dose, followed by a maintenance dose of 1 mg. A 7 fr triple-lumen CVC and the right radial arterial line were given. Two-lung ventilation was conducted till the opening of the hemithorax. A recruitment maneuver was given by applying a pressure of 40 cm water at the end of inspiration for 15 s. Then, the tracheal lumen was claimed. The right lung was allowed to collapse. One lung ventilation was continued with a tidal volume of 270 ml, RR – 14/min, and positive end-expiratory pressure – 7 cm water.
Surgery was done by thoracotomy. Arrow was retrieved en bloc and the right lung was repaired [].
Extubation was uneventful. Spontaneous respiration was observed. Postoperative care included multimodal analgesia with injection paracetamol 1 g TDS, injection tramadol 100 mg BD, erector spine block, and thoracic epidural at the T8 level. Furthermore, nebulization, antibiotics, respiratory, physiotherapy, and incentive spirometry were used.
The patient was discharged after 7 days.


A 53-year-old non-smoking Japanese woman was admitted to our hospital with a 20-year history of wet cough and dyspnoea on exertion. BA had been diagnosed 20 years earlier. Although she has been treated with high-dose inhaled corticosteroid, a long acting β2 agonist and a leukotriene receptor antagonist, she experienced frequent exacerbations of BA, and short-term oral corticosteroid bursts were occasionally administered. Her FeNO was elevated (53 ppb). Blood tests revealed eosinophilia (3480 /µL) and a high total IgE level (353 IU/mL). High-resolution CT (HRCT) of the chest revealed diffuse centrilobular nodules and bronchial wall thickening in both lungs. In addition, patchy ground-glass opacities were observed around the bronchioles (). The pulmonary function test demonstrated obstructive impairment (forced expiratory volume in 1 s (FEV1): 1.55 L, 65.7%; coefficient G for the ratio of FEV1 to forced vital capacity: 57.8%; the ratio of residual volume to total lung capacity (RV/TLC): 43.3% with airflow obstruction). The result of bronchodilator reversibility testing was positive.


A 12-year-old girl was referred to our medical center with a diagnosis of bilateral neuroretinitis and vasculitis. She had no history of previous systemic diseases. An ocular examination revealed a best-corrected visual acuity (BCVA) of 20/20 in the right eye (RE) and 20/40 in the left eye (LE). Intraocular pressure was 12 mm Hg in both eyes; there was no inflammation in the anterior chamber, but mild vitritis, vasculitis, papillitis, and macular exudates in the fundoscopy exam were observed (shown in Fig. ). A fluorescein angiography showed peripheral ischemia and macroaneurysms (shown in Fig. ). Several tests had previously been performed, including a purified protein derivative test which resulted negative; infectious serologies including treponemic serology, Bartonela henselae serology, Toxoplasma gondii serology, and Borrelia burgdorferi serology, all of them negative; brain magnetic resonance imaging and chest X-ray, which were normal; HLA-B51 was positive and antinuclear antibodies at 1/50 titer. There was no history of oral or genital ulcers, joint inflammation, or cutaneous hypersensitivity that were suspicious for Behcet disease. No fever or pulmonary or cutaneous manifestation was present at presentation. Her pediatrician ruled out any systemic autoimmune disease.
Due to the typical clinical findings and because other etiologies were ruled out, a diagnosis of IRVAN syndrome was established. Treatment was initiated with oral corticosteroids of 30 mg at tapering doses, and PRP was performed. Three months later, with the patient off steroids, worsening of BCVA (20/50 and 20/60 in RE and LE respectively) and ME was observed, and it was treated with 2 periocular triamcinolone injections and one intravitreal injection of bevacizumab without success. Hence, a dexamethasone implant was administered in both eyes with improvement in OCT (shown in Fig. ) and slight recovery in BCVA (20/40 and 20/50). The dexamethasone implant was effective, but recurrence of the ME occurred, so during the following 4 years, 9 dexamethasone implants were given in each eye. The patient developed a very mild posterior subcapsular cataract and ocular hypertension in both eyes after the first injection (26 mm Hg), which was well-controlled with timolol drops. During follow-up, control retinographies and fluorescein angiography were performed in order to rule out development of neovascularization, which never happened (shown in Fig. ).
Because of the availability of fluocinolone acetonide intravitreal implant at our institution, we offered the patient this treatment in order to avoid frequent visits and injections. Previous authorization from the pharmacy department of our center was obtained. First, we injected it in her RE to assess effect. The appearance of the macula before the injection is shown in Figure . A decrease in macular thickening was observed at 1 month which maintained at 6 months; at 12 months, a localized perifoveal increase was observed, which was successfully managed with a focal laser. At 2 years of follow-up, BCVA is 20/30 and 20/60 in the RE and LE, the retinal thickening remains stable (shown in Fig. ), and intraocular pressure is 16 mm Hg in both eyes with topical treatment. Remarkably, a decrease in the macular retinal thickening of the LE was also observed after the fluocinolone acetonide intravitreal implant, which has remained until the last follow-up visit (shown in Fig. ). Though retinal thickening has remained stable, recovery of visual acuity has been limited by chronic intra- and subretinal lipidic exudates. Evolution of central macular thickness and VA during the 3 years before fluocinolone acetonide injection (while dexamethasone implants were given) and during the 2 years after this implant was given are shown in Figure .


A 38-year-old man with severe upper abdominal pain presented to our hospital by an ambulance. He had diabetes, atopic dermatitis, depression, and a history of recurrent alcoholic AP. He drank about 90 g of ethanol and smoked 20 cigarettes daily. His vital signs in the emergency room were body temperature (BT) 36.4°C, blood pressure (BP) 143/99 mm Hg, heart rate (HR) 99 beats/min, and respiratory rate (RR) 37 breaths/min. Laboratory tests showed serum amylase 263 U/L, lipase 973 IU/L, C-reactive protein (CRP) 1.42 mg/dL, white blood cell count (WBC) 10,800/μL, total bilirubin (T-Bil) 0.8 mg/dL, aspartate aminotransferase (AST) 57 U/L, alanine aminotransferase (ALT) 67 U/L, alkaline phosphatase (ALP) 106 U/L, γ-glutamyl transpeptidase (γ-GTP) 233 U/L, and lactate dehydrogenase (LDH) 426 U/L. He was diagnosed with alcoholic AP. He underwent conservative treatment. Fentanyl was introduced for pain relief because other analgesics were insufficient. By day 3, >1,200 μL of fentanyl was required daily. At that time, abdominal CT showed marked swelling of the pancreas with extensive inflammation of peripancreatic adipose tissue (Fig. ). His vital signs were BT 38.8°C, BP 133/88 mm Hg, HR 117 beats/min, and RR 42 breaths/min. Laboratory tests showed serum amylase 295 U/L, lipase 581 IU/L, CRP 32.16 mg/dL, WBC 11,200/μL, T-Bil 6.1 mg/dL, direct bilirubin 4.4 mg/dL, AST 178 U/L, ALT 85 U/L, ALP 93 U/L, γ-GTP 323 U/L, and LDH 355 U/L. Sudden onset of jaundice, severe abdominal pain, and high-grade fever suggested the development of coexisting acute cholangitis secondary to an unknown biliary event. Acute cholangitis might have been the main cause of the sudden deterioration of his condition. Therefore, we decided to perform urgent ERCP on day 3. Endoscopy showed a swollen major duodenal papilla (Fig. ) that might occlude the bile duct and the pancreatic duct. We could not detect any biliary stones with cholangiography. Subsequently, EBS and NPD were performed (Fig. ). After drainage, the patient's condition rapidly improved, including a decrease in pain (Fig. ). CT on day 10 showed a dramatic decrease in pancreatic swelling and peripancreatic inflammation (Fig. ). The patient developed pulmonary mycosis and was subsequently treated with antifungal drugs. The causal relationship between pulmonary mycosis and endoscopic treatment was unknown.


A 51-year-old woman presented to our department for treatment of common bile duct stones and liver dysfunction. Endoscopic treatment was performed. That night, she felt severe abdominal pain. Her vital signs were BT 37.7°C, BP 149/98 mm Hg, HR 112 beats/min, and RR 18 breaths/min. Laboratory tests on the next morning showed serum amylase 1,869 U/L, lipase 2,279 IU/L, CRP 0.83 mg/dL, WBC 7,500/μL, T-Bil 2.1 mg/dL, AST 196 U/L, ALT 514 U/L, ALP 384 U/L, γ-GTP 516 U/L, and LDH 235 U/L. Contrast-enhanced CT showed an extensive acute peripancreatic fluid collection, suggesting PEP with biliary obstruction that induced acute cholangitis. Edema at the ampulla of Vater, likely due to the last ERCP procedure, was predicted. She complained of severe continuous pain that required as much as 120 mg of pentazocine over 18 h. We decided to perform urgent re-ERCP for acute cholangitis. Endoscopically, the ampulla of Vater was extremely edematous, which might have occluded both the bile duct and the pancreatic duct. After the second ERCP with EBS and NPD, clinical symptoms resolved rapidly (Fig. ). She did not have any systemic or local complications though previous CT images showed extensive peripancreatic inflammation. Contrast-enhanced CT performed 3 weeks after PEP onset confirmed that there was neither necrosis nor the presence of a fluid collection.


A 52-year-old woman with severe upper abdominal pain and vomiting presented to our hospital by an ambulance. Her vital signs in the emergency room were BT 35.7°C, BP 150/107 mm Hg, HR 87 beats/min, and RR 24 breaths/min. Laboratory tests showed serum amylase 3,045 U/L, lipase 7,000 IU/L, CRP 0.97 mg/dL, WBC 19,200/μL, T-Bil 7.2 mg/dL, AST 472 U/L, ALT 853 U/L, ALP 161 U/L, γ-GTP 434 U/L, and LDH 417 U/L. Contrast-enhanced CT showed an extensive acute peripancreatic fluid collection but no biliary stones. Clinical symptoms and laboratory data led to a diagnosis of obstructive acute cholangitis with a CT-negative biliary stone. We decided to perform urgent ERCP. After EBS and NPD, the severe pain was relieved (Fig. ). Follow-up CT after discharge showed no local complications of AP.


An 82-year-old man presented to our hospital's emergency room with severe upper abdominal pain. He had a history of acute thoracic aortic dissection surgery and cholecystectomy. His vital signs in the emergency room were BT 37.7°C, BP 152/86 mm Hg, HR 112 beats/min, and RR 30 breaths/min. Laboratory tests showed serum amylase 3,592 U/L, lipase 5,256 IU/L, CRP 2.80 mg/dL, WBC 7,100/μL, T-Bil 2.86 mg/dL, AST 382 U/L, ALT 263 U/L, ALP 588 U/L, γ-GTP 929 U/L, and LDH 452 U/L. We decided to perform urgent ERCP because CT and MRI findings suggested a biliary stone impacting the ampulla of Vater. As expected, we found an impacted biliary stone. We removed the stone and performed EBS and NPD. The patient's severe pain resolved after the procedure (Fig. ). The clinical course was good despite the high risk of mortality from AP.


A man in his 40s who had been previously treated with 3 courses of CDDP (75 mg/m2) + pemetrexed (PEM, 500 mg/m2) and 32 courses of nivolumab (240 mg/body every 2 weeks or 480 mg/body every 4 weeks) for recurrent malignant pleural mesothelioma was administered CBDCA (area under the curve 5) + PEM (500 mg/m2) as the third-line treatment. He rarely experienced mild akathisia-like symptoms on his feet (nervous, uneasy, twitchy, restless, and instinct to move) before the diagnosis. The patient stated that he exhibited mild degradation of the symptoms that lasted during CDDP + PEM treatment, which did not require any pharmacotherapy. The patient rarely developed symptoms again during nivolumab treatment. However, the symptoms notably worsened to grade 2 approximately 7 days after the first cycle of CBDCA + PEM and did not disappear. Lorazepam (0.5 mg) was administered 3 times a day from day 14 but was not effective. He complained that the severity of the symptoms was much stronger than that in the CDDP + PEM treatment, and interestingly, symptoms became severe during the day and became milder (almost disappeared) at night. Therefore, we considered that the symptoms were different from restless legs syndrome and that it could be akathisia. However, the patient did not have brain metastasis, infection, or head injury. In addition, his regular medication included 40 mg of the probiotic Clostridium butyricum MIYAIRI, 200 mg of ursodeoxycholic acid, 2 mg of tizanidine, and 2.5 g of Shakuyaku-Kanzo-To, a herbal medicine, 3 times a day, and 10 mg of vonoprazan, 1 g of multivitamin, including 0.5 mg of folic acid, 100 mg of sustained-release ferrous fumarate, and 8 mg of sustained-release hydromorphone, once a day; all of them were previously administered before CBDCA + PEM treatment and did not induce the symptoms. In addition, palonosetron (0.75 mg) and dexamethasone (9.9 mg) infusion on day 1, dexamethasone (4 mg) orally on days 2 and 3, and oral aprepitant (125 mg) on day 1 and 80 mg on days 2 and 3 were administered for chemotherapy-induced nausea and vomiting prevention, which are also considered not to induce persistent symptoms. Consequently, the symptoms continued for 2 weeks from day 7 of the first chemotherapy, and we finally evaluated it to be derived from CBDCA-induced neuropathy as he experienced the same symptoms in CDDP + PEM treatment, although he did not exhibit typical CIPN indications, including numbness, tingling, and pain. Pregabalin is approved for peripheral neuropathy and is suggested to be effective for taxane-induced CIPN and antipsychotic-induced akathisia [, ]. Therefore, we decided to administer 75 mg pregabalin twice daily from the day of the initiation of the second course. As a result, the symptoms were significantly improved by its administration from the day of initiation, which he described as the symptoms having disappeared, even in the daytime, with grade 1 drowsiness. However, he complained that he felt the symptoms again after 10 h from the previous dose, which suggests that pregabalin was effective but its effect weakened or disappeared as time progressed. Chemotherapy was discontinued after the second course, and surgery was performed for disease control. The symptoms gradually improved after completion of chemotherapy.


A 38-year-old man with high myopia and astigmatism presented for a refractive surgery evaluation. The patient had no prior history of glaucoma or ocular trauma. The preoperative refraction was—9.00–2.25 × 85° in the right eye—9.00–3.00 × 85° in the left eye. The patient had a stable refractive history and open angles on gonioscopy. Dilated fundus exam showed myopic changes, tilted discs with a C/D ratio of 0.4 in both eyes. The anterior chamber depth was >3.0 mm and the endothelial cell count was 2,587 cell/mm2 in the right eye and 2,512 cell/mm2 in the left eye with normal morphology. The preoperative evaluation included a comprehensive slit-lamp examination which was unremarkable. The high refractive error and the volume of tissue removal precluded treatment with excimer laser surgery. After a thorough discussion, the patient opted for ICL implantation.
A single PI was performed in each eye at 12 o'clock with a neodymium:YAG (Nd:YAG) laser 4 weeks preoperatively. The Visian ICLs were selected based on sulcus diameters measured by the Pentacam (Oculus Optikgerate GmbH, Wetzlar, Germany).
[−12.00 + 2.00 × 171 OD−13.00–3.00 × 174 OS].
The eye was prepared in a sterile fashion and the two drops of topical anesthetic were placed. A lid speculum placed for maximum corneal exposure. A 3.2-mm peripheral temporal clear corneal wound was created for implantation of the ICL (Visian ICL™ model ICMV4b, Staar Inc., Monrovia, CA, USA). There were no complications during surgery in both eyes.
A few hours after surgery, the patient presented with moderate ocular pain in the right eye, his IOP was 48 mm Hg in the right eye and 19 mm Hg in the left eye (measured with Goldmann applanation tonometry), the anterior chamber was moderately shallow with mild corneal edema. The ICLs were well positioned in the posterior chamber.
The PI was partially patent and the ICLs were in place with a high vault. Retained viscoelastic material was diagnosed. The patient has been prescribed acetazolamide tablets 250 mg qid PO, timolol maleate eye drops (0.5%) bid, and brimonidine tartrate eye drops 0.15% bid. After 24 hours, the IOP decreased to 8 mm Hg and the cornea cleared, so the patient was discharged with instructions to start dilating drops, topical Alphagan (Allergan plc, Dublin, Ireland), and Cosopt (dorzolamide/timolol) 2%/0.5% (MSD, Kenilworth, NJ, USA) for the right eye.
On the second postoperative day, the patient presented with moderate diffuse corneal edema with an IOP of 29 mm Hg in the right eye. The dilating drops were discontinued, augmentation of the PI was performed with a neodymium:YAG (Nd:YAG) laser. A few hours after the YAG PI, the IOP stabilized at 9 mm Hg and the PI was patent but the anterior chamber was relatively shallow, so the dilating drops were prescribed again. Right eye pupillary block was diagnosed. The patient was seen for another two visits postoperatively, with the well-controlled IOP in the right eye; hence, the topical brimonidine tartrate was discontinued.
Four months postoperatively, the patient presented with early lens changes in the right eye (), and IOP was controlled on topical Cosopt. Visual acuity was 20/40 and endothelial cell count was within normal limits. The patient underwent ICL implantation in the left eye and surgery was uneventful. The left eye remained stable for the early postoperative visits. The patient lost to follow-up and presented 4 months after surgery with 20/40 visual acuity in the right eye and 20/60 in the left eye. The IOPs were 19 and 45 mm Hg in the right and left eyes, respectively. The PIs were patent bilaterally with high vault and moderately shallow AC, cataract (), and shallow cupping of the left eye with a C/D ratio of 0.64 and 0.8 in right and left eyes, respectively. The patient was using only Cosopt for the right eye so stat medications were given to reduce IOP in the left eye resulting in a decrease to 22 mm Hg.
Baseline glaucoma investigations indicated a central corneal thickness was 512 and 516 μ in the right and left eyes, respectively. Humphrey's visual field testing (24-2 SITA-standard) was relatively normal in the right eye and showed a superior altitudinal defect in the left eye (). OCT (RTVue; Optovue, Inc., Fremont, CA, USA) imaging confirmed the clinical findings ().
Given the presence of a visually significant cataract and elevated IOP, the phakic ICL in the left eye was removed concurrently with cataract surgery to prevent further visual field loss. Postoperatively, the patient's IOP remained controlled on topical glaucoma medications, and his best-corrected visual acuity improved to 20/20 in this eye.
AS-OCT of the right and left eye () showing an absence of excess ICL vaulting, shallow anterior chamber. Ultrasound biomicroscopy (UBM) demonstrating direct contact (white arrow) between ICL and the posterior iris in the left eye with the shallow anterior chamber and anterior vaulting of ICL consistent with pupillary block following ICL implantation ().


A 2-mo history of recurrent cough, dyspnea, and tachypnea.
A 10-year-old girl was admitted to our unit with a 2-mo history of recurrent cough, dyspnea, and tachypnea. Her condition was more severe after exercise. She had been hospitalized twice in a local unit and diagnosed with bronchitis and asthma. Her symptoms progressed despite treatment with inhaled fluticasone/salmeterol.
The patient had been previously healthy, and she denied a history of asthma and seasonal allergic rhinitis.
The patient had no relevant family history.
On admission, the patient was dyspneic and characterized by biphasic stridor with a percutaneous saturation of 95% on room air. An examination showed a body temperature of 36.7 ºC, a pulse rate of 92 beats/min, a respiration rate of 34 breaths/min, and a blood pressure of 107/74 mmHg.
The patient’s leukocyte count was 6.07 × 109/L (reference range: 4.0-12.0 × 109/L). Her lymphocyte count was 3.09 × 109/L (reference range: 0.7-4.9 × 109/L). An immune function test showed that her immunoglobulins were within the reference range.
Contrast-enhanced computed tomography (CT) revealed an obviously enhanced mass, occluding about 90% of the tracheal lumen (Figure and ). Under conscious and local anesthesia, flexible electronic laryngoscopy showed a red, smooth, and round mushroom-shaped mass. The surface of the mass was covered with several small and discontinuous blood vessels. The base of the mass was in the trachea, about 1 cm below the vocal cords at the 4- to 7-o’clock position (Figure ).
Pulmonary function examinations indicated that the patient, who had exhibited no marked response to bronchodilators, had severe inspiratory obstructive ventilation dysfunction.


A 79-year-old male was admitted to the Department of Gastroenterology of our institution for jaundice that developed 1 mo prior.
The patient had also experienced general weakness and fatigue for a few months before admission, but had shown no change of body weight. The accompanying symptoms were reddish discoloration of urine and pruritus all over the body.
He had taken medicine for hypertension, diabetes mellitus, and benign prostate hypertrophy and had history of tuberculosis pleurisy 18 years before. The patient underwent percutaneous coronary intervention for unstable angina 7 years before. However, the patient had no history of bile duct surgery or intervention.
He was a social drinker and had a 10 pack-year smoking history. There was no family history for autoimmune diseases or biliary cancer.
The patient’s vital signs at admission were within normal limits, with the exception of a body temperature of 38.2 ℃. The skin and sclera showed jaundice. The abdomen was soft and he had no signs of tenderness on the upper abdomen. The gallbladder was not definitely palpable.
Blood analysis revealed no leukocytosis (7560 white blood cells/µL) with normal hematocrit and platelet count. Total bilirubin was markedly increased (17.9 mg/dL; normal range: < 1.2 mg/dL) along with aspartate aminotransferase/alanine aminotransferase (97/52 IU/L; normal range: 4-40/4-41 IU/L) and alkaline phosphatase (339 IU/L; normal range: 35-105 IU/L). Serum amylase/lipase was normal and C-reactive protein was slightly increased (1.48 mg/dL; normal range: < 0.5 mg/dL). Carbohydrate antigen 19-9 was greatly increased (1061 U/mL; normal range < 37 U/mL). Carcinoembryonic antigen was within normal range. Serum IgG/IgG4 levels were not measured. Urine color was dark brown, and bilirubin was 4+ mg/dL.
Computed tomography (CT) scan and magnetic resonance cholangiopancreaticography (MRCP) revealed a short segmental concentric wall thickening of the proximal to mid CBD with diffuse dilatation of the bile duct to the periphery and gallbladder distension (Figure ). Subsequently-performed endoscopic retrograde cholangiopancreaticography (ERCP) demonstrated a 1 cm-long segmental stricture at the proximal CBD level (Figure ), but no malignant cells or specific histopathological confirmation were proven in biopsy tissues of the bile duct. Positron emission tomography (PET)-CT supported these findings by showing a focal hypermetabolic lesion (SUVmax 4.2) in and around the proximal CBD area (Figure ). These findings made a straightforward impression of proximal CBD cancer.


A 75-year-old male without known coronary artery disease presented to the emergency department with acute-onset chest pain. His medical history was significant for recurrent epistaxis. Physical examination was normal except for superficial lip TeLs []. Vital signs included a heart rate of 67 beats/min, blood pressure of 159/80 mmHg, respiratory rate of 18 breaths/min, and oxygen saturation of 87% when breathing ambient air. The complete blood count revealed erythrocytosis with a hemoglobin of 16.9 g/dL and hematocrit of 50.4. Electrocardiogram demonstrated nonspecific ST segment changes in the inferior leads and serial cardiac troponin peaked at 40 ng/ml. Immediate medical therapy included aspirin 325 mg in combination with ticagrelor 180 mg, intravenous heparin infusion, atorvastatin 80 mg, and metoprolol 12.5 mg. Coronary angiography revealed significant multivessel disease. Echocardiogram showed a left ventricular ejection fraction of 50% without evidence of pulmonary hypertension (PH); however, inferolateral hypokinesis and moderate ventricular hypertrophy were noted. A nonemergent coronary artery bypass graft (CABG) was recommended and preoperative testing was performed.
In preparation for CABG, bilateral carotid ultrasound did not indicate significant atherosclerotic disease. A chest X-ray showed several nondiagnostic pulmonary nodular densities which were identified by a contrast-enhanced chest computed tomography (CT) as pulmonary AVMs (PAVMs). Three-dimensional reconstruction provided detailed configuration and distribution of the PAVMs [-]. The patient recalled hemoptysis and hemorrhagic strokes involving both his father and brother. Pulmonary function test revealed normal spirometry and lung volumes and a reduced carbon monoxide diffusion capacity. The periodic hypoxia captured on pulse oximetry was verified with arterial blood gas analysis, which demonstrated a pH of 7.44, pCO2 of 30 mmHg, pO2 of 54 mmHg, and O2 saturation of 89%. The calculated alveolar–arterial gradient (A-a gradient) was elevated at 58 mmHg. Causes of increased A-a gradients include ventilation/perfusion mismatch (i.e., chronic obstructive pulmonary disease [COPD] and pulmonary embolism), diffusion limitations (i.e., pulmonary fibrosis and interstitial lung disease), and both physiologic and anatomic shunts such as atelectasis or pulmonary edema and intracardiac or pulmonary vascular shunts, respectively. Despite supplemental oxygen, the hypoxemia failed to improve, highlighting a unique feature of the right-to-left shunt that accompanies PAVMs. Immediate intervention of the PAVMs was postponed and CABG prioritized. Brain magnetic resonance imaging (MRI) to screen for additional AVMs was negative; however, several bilateral chronic lacunar infarctions were detected in the cerebellum.
Postoperatively, aspirin 325 mg daily, atorvastatin 40 mg daily, and cardiopulmonary rehabilitation were initiated; however, beta-blocker and ACE inhibitor were postponed due to the development of sinus bradycardia and acute kidney injury. New-onset atrial fibrillation with tachycardia occurred on postoperative day 3 and therefore apixaban 5 mg twice daily and metoprolol tartrate 12.5 mg twice daily were commenced. Following his discharge, physical rehabilitation and outpatient cardiology evaluation were maintained.
Three weeks following discharge, he returned to the emergency department with symptoms of blurry vision and an unsteady gate. Vital signs were normal and electrocardiogram demonstrated normal sinus rhythm. A noncontrast CT and contrast-enhanced MRI of the brain revealed an evolving subacute stroke with microhemorrhages in the right occipital lobe []. Multiple imaging studies for an embolic source included carotid magnetic resonance angiography (MRA), repeat echocardiogram, and lower extremity venous duplex which were negative. MRA of the brain to assess for an aneurysm or clot amenable to endovascular intervention was negative; however, multifocal segmental narrowing of the right posterior cerebral artery was present []. The symptoms subsided, and following discharge, evaluation by interventional radiology recommended PAVM embolization and genetic testing to allow accurate testing of family members. The patient was also referred to gastroenterology to undergo screening for possible abdominal–visceral AVMs.


An 18-year-old male presented to the Eye Emergency Department at a tertiary eye centre complaining of a 2-day history of blurred vision in the left eye accompanied by mild redness and aching. There was no watering, discharge, photophobia, floaters or flashes, and he was asymptomatic with respect to his right eye. The patient denied any foreign body or other recent ocular injury, nor any similar preceding episodes. Review of systems was unrevealing, in particular he denied rashes, cold sores, malaise or recent intercurrent illness. His past ocular history and past medical history were similarly unremarkable.
The best-corrected visual acuity was measured at 6/5 in both eyes despite subjectively blurred vision on the left. A slit-lamp biomicroscopy showed the right eye was normal. The left eye had ciliary injection with signs of anterior chamber inflammation. 1 + cells with minimal flare was observed, as were numerous fine keratic precipitates (KPs) on the inferior corneal endothelium of the left eye. Both anterior chambers were deep and no notable abnormalities of iris or vitreous were observed in either eye. The IOPs were recorded as 18 mmHg in the right eye and 35 mmHg in the left (normal range < 21 mmHg) by Goldman applanation tonometry.
On funduscopy, spontaneous retinal arterial pulsation could be seen at the disc in the left eye, as per the . There was no corresponding pulsatile activity in the right eye, and no spontaneous venous pulsation (SVP) could be seen in either eye.
The clinical impression was of acute anterior uveitis with relatively modest anterior chamber inflammation, complicated by raised IOP. There were no signs clearly indicating any underlying aetiology to the uveitis, and it was therefore presumed to be viral in nature, viral uveitis being a frequent cause of uni-ocular hyptertensive uveitis. The patient responded well to topical pressure lowering agents, with an IOP measuring 18 mmHg 1 hour after administration. Typically, these cases are highly responsive to topical steroids, and therefore, prednisolone acetate 1% drops were given four times daily. Antiviral agents were not given in the absence of granulomatous or severe inflammation or corneal involvement. The patient was discharged with regular Dorzolamide + Timolol drops to the left eye to control the IOP, a tapering course of topical steroid and a short course of topical cycloplegic therapy for comfort. Review was scheduled in clinic over the following weeks.
The patient did not attend initially scheduled follow-up; however, he was seen some months later in clinic. At this point, he reported rapid resolution of his symptoms with complete return to subjective normality of his eyes. Clinical examination confirmed entirely normal findings with full visual function. He was subsequently discharged and has not re-presented to our eye service.


A 50-year old healthy man presented with a brown irregular flat lesion surrounding the tip of the glans, involving the urethral meatus (). The lesion biopsy showed melanoma in situ. Urethrocystoscopy revealed that the melanoma was spreading into the fossa navicularis with less than 1 cm infiltration. The staging with total-body computed tomography (CT) scan did not reveal distant metastasis. After multidisciplinary discussion, radical surgery was planned including the removal of the glans and distal urethra.
At the first-stage surgery an anatomic total glansectomy was performed, exposing the intact tips of the corpora cavernosa. The distal urethra was excised with a 1.5 cm safety margin, confirmed as healthy by a preliminary histological examination (). A dorsal transverse preputial flap was created, and a template-tailored free graft was harvested from the internal leaflet of the foreskin (). The graft was accurately meshed and sutured with 4-zero polyglactin stitches upon the ventral aspect of the corpora cavernosa, laid on the bed where the removed urethra was previously situated. Subsequently, a free full-thickness skin graft of adequate size was harvested from the hairless area of the left groin, being suitable for use as a patch to restore the aesthetic aspect of the dorsal dome of the corpora cavernosa. The skin graft was prepared separately by accurate defatting until achieving a thin thickness, meshed with scalpel, laid circularly for 180 degrees on the tip of the corpora cavernosa, and sutured with 4-zero polyglactin interrupted stitches (). Medication with embedded gauzes was left inside for 3 days, followed by office medications until complete healing. The final pathology examination confirmed the melanoma in situ with free margins.
The second-stage surgery was planned after 6 months at consolidated first-time reconstruction (). The plate of preputial graft was incised along its edges and tubularized using a running 5-zero polyglycolic suture upon a 14 Fr catheter (). A dartos tail was used to cover the neourethra to protect the suture, and then the shaft was covered with a tension-free penile skin flap (). Urethral catheter was removed in three weeks, after a para-catheter retrograde urethrography confirming the watertight suture profile.
At 24 months after the second-stage surgery, the urethra was shown to be negotiable by a 16 Fr catheter (). Voiding function recorded a normal peak flow, including linear and turbulent micturition. Erectile function was normal. The poor skin sensitivity on the resurfaced area of the corpora cavernosa had significantly improved at the last follow-up visit (the patient self-reported sensitive evaluation was ‘good’, while 1 year before, it was ‘bad’). No local disease recurrence was observed and the whole-body CT scan was negative.


A 16-year-old girl presented with slowly progressive headache. MRI showed a well-defined, 14×16×20 mm, intradural extramedullary cystic lesion in the premedullary cistern (). The lesion was homogeneously hyperintense on T1- and T2-weighted images. There was no restriction on diffusion-weighted imaging. Slightly elevated cerebral blood flow and cerebral blood volume were noted in the anterior part of the cystic mass on perfusion-weighted imaging. There was no gadolinium contrast enhancement on MRI.
We decided to perform surgery to prevent symptom progression and avoid neurologic deficit. A far-lateral transcondylar approach was chosen to secure the surgical field and prevent cranial nerve injury (). We performed lateral suboccipital craniotomy with minimal condylectomy and secured the surgical field, which included the anterior aspect of the medulla and the premedullary cistern. The cyst was located following arachnoidal dissection of the cranial nerves and vertebral artery. The cyst was covered by a light gray capsule, which was loosely connected to the brain parenchyma. The cystic wall was punctured, and an amorphous gray-yellowish fluid was drained. The residual cyst capsule was completely detached from the adjacent neurovascular structures. Histopathological examination revealed non-ciliated, mucin-producing columnar and cuboid epithelial cells with isolated goblet cells, which are characteristic features of neurenteric cyst (). Complete surgical resection of the cyst was performed without any complications. MRI conducted postoperatively confirmed that the mass had been completely resected (). No adverse events occurred during 6-month follow-up period.


A 38-year-old white male, accountant, known to have diabetes, hyperparathyroidism and chronic renal failure on regular hemodialysis for five years, he was presented to our emergency department with history of minor fall while he was going down stairs, he had severe bilateral knees pain and inability to ambulate.
Clinical examination showed bilateral knees massive effusion, right infra-patellar ecchymosis and left supra-patellar obvious depression. There was palpable soft tissue defect in the right patellar tendon and the left quadriceps tendon, restricted both knees range of motion with extreme patellae mobility and he was unable to straight raising his both legs. Plain radiographs showed right Patella Alta and left Patella Baja, but there was no evidence of patella fracture or any calcified deposits in either quadriceps tendons or patellar tendon (). Magnetic Resonance Images (MRI) on sagittal plane T2-weighted of right knee showed complete transection of the patellar tendon from its origin with superior displacement of the patella, associated with infra-patellar large hematoma, thickened edematous quadriceps tendon and disruption of the patellar retinaculum; while left knee showed complete transection of the quadriceps tendon at its insertion with inferior and lateral displacement of patella associated with a supra-patellar large hematoma, thickened wavy patellar tendon and disruption of the patellar retinaculum ().
The patient underwent bilateral quadriceps and patellar tendon open surgical repair. Right knee infra-patellar longitudinal skin incision was made, the inferior pole of the patella and patellar tendon were exposed. Intra-operatively, there was total rupture of the patellar tendon at its origin with distal tendon retraction, thorough washing and debridement of unhealthy patellar tendon stump, bony refreshment of inferior pole of the patella and retracted patellar tendon was sutured to the distal pole of patella with two non-absorbable heavy anchor sutures using a Krackow’s suture technique. Left knee supra-patellar longitudinal skin incision was done. Exposure of the quadriceps tendon along with the proximal patella was done. Intra-operatively, there was total rupture of quadriceps tendon at its attachment to the superior pole of the patella, thorough washing and debridement of unhealthy quadriceps tendon stump, bony refreshment of superior pole of the patella and retracted quadriceps tendon was sutured to the proximal pole of patella with two non-absorbable heavy anchor sutures using a Krackow’s suture technique. Both repaired tendons were stable intra-operatively during stressing with passive knee flexion. Post-operatively, patient was placed in bilateral hinged knee brace of 0–30°, then he started physiotherapy within few days after surgery, partial weight bearing mobilization was allowed using walking crutches, gradual increase of knee flexion within brace was applied. The patient used the knee braces and the walking crutches for two months until complete tendons healing with sufficient strength in his repaired tendons. At four-year follow-up, radiographs confirmed solid healing of all repaired tendons. Patient had a satisfactory clinical outcome. His functional range of motion of both knees was near normal, 0°-100° ().


A 67 year-old woman was referred to our hospital’s oncology clinic for the management of a skin-tumor in the left cervical area who underwent surgical resection twelve months before, at a primary care facility. The histology exam she provided described a mixed basal cell and squamous cell carcinoma.
Histological re-evaluation of the paraffin bloc performed in our department of pathology revealed a tumor growth with focal ulcers on its surface, with a solid architecture, cell nests or thick trabecula, some emerging from superjacent epidermis and infiltrating the profound reticular dermis. The tumor was constituted of pleomorphic cells, with round-oval nuclei, some with prominent nucleolus, dispersed chromatin, amphophilic or eosinophilic cytoplasm, with evident cell limits. Small cystic structures were observed focally. Mitotic activity was high – 4-5 mitoses/high power field – and the tumor stroma was fibrous and in low quantity. A moderate lympho-plasmocytic inflammatory reaction was observed around the tumor. The profound dermal and hypodermal layer contained satellite tumor nodules with focal necrosis, away from the tumor invasion front (). Immunohistochemistry staining of the tumor cells revealed diffuse positivity for p63, zonal positivity for epithelial membrane antigen (EMA) and the low molecular-weight cytokeratin CK7. Carcinoembryonic antigen (CEA) was present in rare tumor cells and the B-cell lymphoma-2 marker (Bcl-2) was positive in some of the tumor cells. Estrogen-receptor was weakly positive in rare neoplastic cells, while the tumor was negative for Gross cystic disease fluid protein 15 (GCDFP15). Immune-histo-chemical analysis is summarized in . The histology diagnostic was an eccrine porocarcinoma.
The patient was re-evaluated with thoraco-abdomino-pelvic CT-scan, which excluded secondary lesions. On the other hand, the cervical CT-scan revealed multiple left lateral cervical lymphadenopathies moderately enhancing after contrast and two other left jugular-carotid lymphadenopathies. After multidisciplinary discussion, 4 cycles of chemotherapy with platinum derivatives and taxanes were administered, after reviewing the literature [, , ]. However, cervical CT-scan performed two months later showed an important increase in contrast enhancement of one of the left lateral cervical lymphadenopathies, with stationary imaging aspects of the other previously described lymphadenopathies (). Given this increased size of cervical lymph nodes, the patient was considered a non-responder to chemotherapy, and surgery was indicated. The patient underwent a wide resection of the previous operative-scar with left latero-cervical lymphadenectomy of the superior compartment (IIa). The cutaneous surgical specimen was tumor free and all of the eleven harvested lymph nodes were negative for metastasis on the histology exam.
Three months later, as part of the patient’s follow-up, a cervical soft-tissue echography revealed a left supero-lateral cervical hypoechogenic lesion, 9/12/9 mm in size, with Doppler signal present inside, well-defined in contact with the inferior pole of the parotid gland. Two other satellite nodules with similar characteristics were observed, one at the superior pole of the lesion and another at the inferior pole of the lesion, measuring 4 mm and 3.8 mm respectively. No lateral cervical lymphadenopathies were observed on either side. The right parotid gland was normal as well.
A positron emission tomography (PET) scan with fluoro-deoxy-glucose (FDG) performed 6 months after surgery described an intense metabolic activity of a left lateral cervical nodular lesion, 14/15/27 mm in size, in contact with the inferior pole of the left parotid gland and the anterior aspect of the left sternocleido-mastoid muscle. No pathological metabolic activity was observed on the lateral cervical nodes bilaterally and the rest of the body. The patient underwent surgical excision of the adenopathy under the parotid gland. Histological examination confirmed the malignant nature of the adenopathy, describing a nodule with extensive metastasis from a carcinoma with solid-type insular architecture, with necrosis in certain comedo-type tumor islets. Mitotic activity was evaluated at 7-8 mitosis / high-power field ().
The patient underwent postoperative external radiotherapy 50 Gy/25 fractions for CTV-N-LR (low-risk nodal clinical target volume) - ipsilateral neck levels Ib, II, III, IV, V, VIII, 60 Gy/30 fractions for CTV-N-HR (high-risk nodal clinical target volume) - ipsilateral neck levels II, VIII. Cervical CT-scan after radiotherapy revealed no signs of tumor lymphadenopathies or distant metastasis which, according to the data from literature [, ], this would indicate a good long-term prognosis.


A 32-year-old man with a history of drug abuse presented to the emergency department with deterioration of paresthesia and limited movement of the left neck for 7 d.
The patient had slight paresthesia of the neck for 5 years. Seven days before this admission, the symptom became intolerable and was accompanied by a limited movement of the neck after an acute upper respiratory tract infection. He perceived a foreign body existing on the left side of the neck and the feeling was severe while coughing or swallowing.
The patient had no history of neck trauma, cervical spondylosis, or contagious diseases.
The patient was a heroin abuser 5 years ago and achieved successful detoxification for 2 years. He had no family history of hereditary diseases.
There was no abnormality in vital signs. Physical examination revealed only mild pain during the palpation of the left neck, and a rod-shaped solid substance was touched. Its location was parallel with the long axis of the left cervical artery.
A blood test revealed a slightly elevated C-reactive protein level (1.53 mg/dL) and white blood cell count (10.94 × 109/L). Other blood test results were normal.
The radiological examination showed that a rod-shaped foreign body was diagonally located in the anterior region of the neck and the lower end of the rod extended to the thorax (Figure ) and computed tomography revealed a high density extending from the cervical region to the upper mediastinal area (Figure ).
Because of the unexpected imaging findings, further medical history was acquired. He finally admitted that he deliberately swallowed a segment of mercury thermometer after he broke a mercury thermometer and poured out the mercury 5 years ago under the influence of drugs when he was alone. He was sent to the emergency department immediately to undergo an endoscopy after he told his family about the accident. But neither a foreign body nor trauma or perforation was found. He did not receive further examinations or treatments because the clinician thought that he was talking nonsense under the influence of drugs, and the patient’s symptoms were tolerable until 7 d before this admission.


In April 2012, a 67-year-old Chinese woman was initially found to have an CA19-9 of 57 U/mL (normal range 0-35 U/mL) and experienced a symptom of left back pain.
The patient denied having left back pain, CA19-9 elevation or other clinical manifestations.
The patient had a clean medical history.
There was no family history of cancer.
No abnormality was found upon physical examination.
Blood analysis showed that CA19-9 was elevated to 57 U/mL (normal range 0-35 U/mL).
An initial imaging evaluation with a thoracic-abdominal computed tomography (CT) scan revealed a mass located in the pancreas.


A 64-year-old postmenopausal female, P5 L5A1, attended a gynecological clinic with a history of mucoid to foul-smelling white discharge per vagina for the past 6 months. Her previous menstrual cycles were regular, the perimenopausal transition was smooth, and she attained menopause 14 years ago. She was previously asymptomatic with no history of postmenopausal bleeding. She had five spontaneous vaginal deliveries and one termination of pregnancy (medical termination of pregnancy [MTP]). Her antepartum and postpartum periods were uneventful with five living issues and her last childbirth 35 years back. Thirty years back, she underwent MTP at 16 weeks by dilatation, evacuation, and history of excessive bleeding, requiring blood transfusion. No other history of past surgeries or tuberculosis. She is a well-controlled hypertensive on medication and coronary artery disease, for which she underwent percutaneous transluminal coronary angioplasty and stenting 5 years ago.
On examination, general physical examination was unremarkable. The abdomen was soft, with no mass or organomegaly. On gynecological examination, the vulva and vagina were atrophic. Per speculum revealed pus-like discharge with normal ectocervix. The uterus was enlarged to parous size, mobile with free fornices. Pap smear was inflammatory and negative for intraepithelial lesion or malignancy. Her ultrasonography revealed an enlarged uterus of 10.7 cm × 7.5 cm filled with linear, echogenic structure in the endometrial cavity up to the cervix with fluid around. The endometrial cavity showed evidence of pyometra. Differential diagnoses on ultrasound were suspected retained bones or dystrophic calcification. Magnetic resonance imaging done showed evidence of pyometra of 39 mm × 26 mm × 31 mm and bone-like echogenicity in the endometrial cavity. The patient underwent hysteroscopy, which revealed multiple pieces of shredded bony structures embedded in the endometrial cavity, all removed using forceps []. The endometrial cavity did not have any abnormal vascularity or hyperplastic changes. Endometrial sampling was done, and specimens were sent for histopathological examination.
The microscopic examination of the paraffin-embedded H and E sections showed woven bone in bony bits, and endometrial tissue showed foamy histiocytes with multiple tiny calcareous round basophilic bodies (Michaelis-Gutmann bodies) along with lymphocytes, hemorrhage, and fibrin []. Von Kossa and periodic acid Schiff (PAS) stain highlighted the basophilic bodies; Perl's stain highlighted the iron pigment []. Histopathological diagnosis of malakoplakia with osseous metaplasia was made.


An 18-year-old male was admitted to the department of neurosurgery on March 14, 2016, with headache, nausea, and vomiting for 2 days. The patient denied a history of head trauma, operation, or any other infectious or systemic diseases, and he was not taking any medication.
On examination, the patient’s consciousness level according to the Glasgow Coma Scale was E4V4M6, and the patient exhibited confusion, lethargy, and disorientation. There were no meningeal signs or any other neurological deficits. The temperature on admission was 36.8°C, and the blood pressure and laboratory tests were within the normal range. A noncontrast computed tomography (CT) scan of the head showed a mixed density lenticular mass with some air collection in the left frontal region [], which is associated with compression of the left lateral ventricle, and a 10 mm midline shift to the right and effacement of the left ambient cistern were observed []. The bone window revealed thickening of the left frontal bone []. The axial sinus CT image demonstrated opacification of the left frontal, ethmoid, and maxillary sinuses [], and the corresponding image in the bone window revealed diffuse thickening and sclerosis of the right maxillary sinus wall [], suggesting paranasal sinusitis and osteomyelitis of the maxillary wall.
After obtaining informed consent for surgery, an emergency operation was performed. Under general anesthesia, the patient was placed in a supine position, and a left frontotemporal craniotomy was performed. When the first burr hole was made, approximately 50 mL of odorous, thick, yellow pus was removed from the epidural space and bone edge. After elevation of the bone flap, bone was further removed until the normal bone edge was encountered. The infected bone was hypertrophic and soft, and its surface was irregular with some small pores. A fresh EDH beneath the abscess was also found after removal of the EDA, which was carefully evacuated. After abundant irrigation, subdural exploration was deemed unnecessary. The bone flap was discarded. Before closure, the wound was thoroughly washed with povidone-iodine and hydrogen peroxide, and an epidural drainage tube was placed.
On the 1st day after the operation, a CT scan revealed that the left frontal EDA was removed, with return of the midline and enlargement of the ambient cistern []. The epidural drainage tube was removed 48 h after surgery. The pus was examined for fungi, Mycobacterium tuberculosis, and aerobic and anaerobic bacteria, but no organisms were identified. Histopathological examination of the bone flap revealed findings consistent with chronic osteomyelitis, including inflammatory granuloma, massive bone necrosis, and pus formation, but no neoplastic process was identified. The blood culture was also negative. Echocardiography did not show any signs suggestive of endocarditis.
Empiric antibiotic therapy (vancomycin 1 g bid combined with metronidazole 500 mg bid) was initiated intravenously for 4 weeks. Postoperatively, the patient’s condition improved with the resolution of symptoms, and he was discharged home 4 weeks later with an indication for 4 more weeks of oral antibiotic therapy (cefixime capsule 100 mg bid). A subsequent CT scan 3 months after surgery showed no signs of infection [-]. Cranioplasty with titanium mesh was performed nine months after surgery [], and the patient recovered uneventfully. At the 3-year follow-up after cranioplasty, the patient was doing well.