A 66-year-old woman visited our clinic with a history of loose stools without anal symptoms. Her past medical history and family history were unremarkable. The laboratory blood tests, including white blood cell count, hemoglobin, platelets, alanine aminotransferase, and aspartate transaminase, were within the normal limits. A viral marker for hepatitis B and a serologic investigation for the immunocompromised state (HIV antibody) were negative. The patient underwent a colonoscopy which revealed an approximately 5-mm-sized, yellowish, submucosal tumor in the anal canal (). On digital rectal examination, a 5-mm-sized, hard, movable tumor was palpated in the anterior wall of the anal canal. The patient was anesthetized with a caudal block, and the tumor was excised using a transanal approach. The histopathology of the specimen revealed AGC, and microscopic examination showed a cystic lesion lined by squamous epithelium and glandular tissue composed of columnar epithelium in the lumen (). The patient’s postoperative course was uneventful, and she was discharged on postoperative day 3. No recurrence was observed after 6 months of follow-up.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. This study was approved by the electronic Institutional Review Board of the Korea National Institute for Bioethics Policy (KoNIBP) (IRB No. P01-201807-21-012).


A 23-year-old male was presented to the out-patient department of our institute with complaints of dyspnoea (NYHA class –II) for last one year. No history of orthopnoea or paroxysmal nocturnal dyspnoea was present. The patient also gave a history of regular palpitations on exertion for the last five years for which he did not seek any medical consultation. There was no history of frequent chest infections in childhood. No history of cyanosis, abdominal fullness, or pedal edema was present. On examination, his blood pressure was 142/56 mmHg, pulse rate was 92bpm and respiratory rate was 16 /minute. Oxygen saturation on room air was 92%. On cardiovascular examination, there was grade 3 parasternal heave with palpable P2. On auscultation, P2 was loud.
There was a grade 3/6 systolic murmur over the left parasternal area in 2nd and 3rd intercostal space and another pan-systolic murmur over lower left parasternal area i increasing with inspiration suggestive of tricuspid regurgitation.
Electrocardiography (ECG) showed normal sinus rhythm with features of biventricular hypertrophy. Chest x-ray posteroanterior view (Fig. ) showed cardiomegaly with down and out apex (suggesting left ventricular pattern of enlargement). Main pulmonary artery (MPA) was enlarged with dilated right descending pulmonary artery (measuring ~17mm). In addition, there was a paucity of vascular markings in outer one-third of lung fields bilaterally. On transthoracic echocardiographic examination, there was Left Ventricular (LV) enlargement (LVEd: 79mm) with normal LV systolic function (LV ejection fraction: 62%, measured using modified Quinones equation by M-mode) and mild tricuspid regurgitation (TR gradient: 92mmHg). There was turbulence in MPA with the presence of AP window (defect size: 29 mm) in parasternal short axis view (Fig. . Colour Doppler showed the presence of left to right shunt (Fig. ) with a peak gradient of 45 mmHg. No other associated cardiac anomaly was found. The patient subsequently underwent Computed Tomography (CT) pulmonary angiography, which confirmed the diagnosis of AP window (Fig. ). The defect measured ~32mm anteroposteriorly and ~28mm craniocaudally, and was located midway between semilunar valves and bifurcation of the main pulmonary artery. MPA was dilated, measuring ~38mm. Right, and left pulmonary arteries were also dilated (measuring ~25mm and 24mm, respectively). In addition, first and second order intraparenchymal branches of pulmonary arteries were also dilated with the pruning of distal branches (Fig. ). Patchy areas of mosaic attenuation were also observed in bilateral lungs (Fig. ) suggestive of perfusion related changes.
A final diagnosis of AP window with severe PAH was made. The patient was advised catheterisation study to rule out irreversible PAH (Eissenmenger syndrome), as it is a contraindication for corrective surgery. However, the patient refused catheterisation study and requested a referral to a higher center due to the non-availability of corrective surgery in our institute. Unfortunately, the patient was lost to follow up after the referral.
Video: Parasternal short axis view on transthoracic echocardiography demonstrating abnormal communication (white arrow) between ascending aorta (Ao) and main pulmonary artery (MPA). (A higher resolution / colour version of this figure is available in the electronic copy of the article).


A 52-year-old male presented with 1-year history of bilateral lower extremity radiculopathy with paraesthesia extending to the toes. The neurological examination revealed bilateral lower extremity hyperreflexia and Babinski responses plus a 10% loss of light touch in the left S1 distribution.
CT and MR examinations showed an abnormal bony mass arising from the posterior arch of T10 that protruded into the spinal canal, resulting in marked canal stenosis and cord compression. The cortex and medullary portions of the lesion were continuous with the T10 vertebral body (i.e., exostosis) []. The MR T1- and T2-weighted images revealed a hypointense lesion occupying the left posterolateral spinal canal at the T10 level with accompanying posterior cord compression. Further, there was a hyperintense signal on T2/STIR images within the cord itself consistent with edema [ and ].
Surgery consisted of a bilateral T10 laminectomy for en bloc resection of the lesion, a predominantly bony mass originating from the left lamina of T10 that was (e.g., continuous with the inner surface of the T10 lamina on the left) []. Postoperatively, the patient immediately recovered normal neurological function.
The histopathologic revealed a bony lesion with a cartilaginous cap covered with perichondrium, endochondral ossification continuous with bony trabeculae, and benign chondrocytes consistent with the diagnosis of osteochondroma [ and ].


A 36-year-old otherwise healthy male presented to an outside hospital with blurred vision for few days and acute onset of aggressive behavior and agitation. While in the emergency department, he deteriorated rapidly to a Glasgow Coma Scale of 8 requiring intubation. A head computed tomography (CT) revealed a large, bifrontal extra-axial mass of the anterior skull base, measuring 52 mm × 37 mm. The lesion also involved the ethmoid cells and frontal sinuses as well as the right orbit. It was infiltrating the dura. Extended peritumoral brain edema with significant mass effect was also noted []. The patient was emergently transferred to our hospital. On arrival, he had fixed mid-dilated pupils, with minimal flexion movement to pain and intact corneal and gag reflexes. Laboratory tests were within normal limits. He was treated with intravenous push of high- dose dexamethasone (20 mg) and mannitol (70 g) and was immediately transferred to the operating room for a bifrontal decompressive craniectomy and tumor debulking.
At surgery, the tumor was found to be very vascular. On opening the dura, the brain was swollen and of firm consistency with no pulsations. A debulking operation was performed along with bifrontal decompressive craniectomy. An intracranial pressure (ICP) monitor was inserted to the parenchyma. The ICP levels were <25 mmHg on average during the postoperative course, the patient was in Glasgow Come Scale 3, with no brain stem reflexes. Postoperative head CT demonstrated persistent, bilateral, massive edema, the patient’s condition remained critical and he passed away 1 week later.
Histological examination for hematoxylin and eosin stain showed a tumor comprised small to medium size cells, some of them with clear cytoplasm. Rhabdoid cells were not noted. The tumor shows areas of necrosis and high levels of mitotic and apoptotic activity [ and ]. The stain for INI1 was negative (loss of expression) []. Immunohistochemical stains were diffusely and strong positive for epithelial membrane antigen (EMA) [] and P63, partially positive for smooth muscle antigen (SMA) [], glial fibrillary acidic protein (GFAP) [], B-cell lymphoma 2, and vimentin (VIM), focally positive for pan-KER and KER 8, and negative for OLIG2, S100, desmin, synaptophysin, NEU-N, and chromogranin. The proliferative index Ki-67 was 25–30%. The initial diagnosis was ATRT. Later, the possibility of SMARCB1-deficient sinonasal carcinoma was added as a differential diagnosis.


A 49-year old woman was referred to our hospital for a small colonic subepithelial tumor (submucosal tumor) detected by screening colonoscopy. Mucosal biopsy specimens from other clinics showed no specific findings. She had no significant medical history or abdominal symptoms. The physical examination and laboratory findings were normal.
A yellowish protruding lesion about 1 cm in diameter was observed in the hepatic flexure on colonoscopy. The surface mucosa was normal, and the lesion showed a hard consistency. The pillow and rolling signs were positive. These findings were suggestive of a subepithelial tumor. (). EUS was performed for the differential diagnosis of a submucosal tumor and visualized a 1-cm sized homogenous hypoechoic lesion arising in the second and third layer of colonic wall, with the first layer preserved (). There was post-acoustic shadowing of a slightly hyperechoic foci inside the lesion (red arrow). Due to the small lesion size, it was difficult to perform EUS-guided fine-needle aspiration (EUS-FNA), and premalignant subepithelial tumors such as neuroendocrine tumors could not be excluded. The patient had psychological anxiety regarding the unclear tumorous lesion. We decided to perform ESD for diagnostic treatment. Hypertonic saline-epinephrine solution was injected into the submucosa to elevate the lesion. A dual knife (KD-650U; Olympus, Tokyo, Japan) was used for dissecting the submucosa. The lesion was completely resected, and no residual lesion remained (). The tumor was well-demarcated and whitish. Deep mucosal injury occurred during the procedure and an oozing visible vessel was observed. Five hemoclips were used for hemostasis, and bleeding was successfully controlled. There were no specific complications after the procedure, and the patient was discharged the next day.
Histologically, a relatively clear, unencapsulated mass was observed. Hematoxylin and eosin stained specimens showed hypocellular spindle cell proliferation, abundant hyalinized collagen, and prominent lymphoplasmacytic inflammatory infiltration. Immunohistochemical staining for Desmin, C-kit, and S100 showed negative results. These histopathologic findings were consistent with CFT (, ).


A 28-year-old woman was referred to our department with a complaint of blurred vision during a period of 1 month in her right eye (RE). She reported nyctalopia and restricted visual field in both eyes since childhood and had been diagnosed RP with no family history. She also had had bilateral hearing impairment since childhood and wore hearing aids.
At presentation, her best-corrected VA was 2.5/10 (− 1.00sph) in the RE and 3.5/10 (− 1.00cyl × 90°) in the left eye (Snellen). Anterior segment examination was unremarkable with bilateral clear lenses. Dilated fundoscopy revealed a full-thickness macular hole and posterior vitreous detachment in the RE. In addition, both eyes showed typical signs of RP including peripheral retinal atrophy with bone spicule-shaped pigmentation in the mid periphery, waxy pallor of the optic nerve head and attenuation of retinal vessels (Fig. ). Spectral domain optical coherence tomography (SD-OCT) confirmed the presence of a FTMH stage IV in the RE along with atrophy of the outer retinal layers and absence of the ellipsoid zone (Fig. a). In the fellow eye, thinning of the outer retinal layers was also observed, while the ellipsoid zone was only preserved in the fovea (Fig. b, d, f).
The patient underwent 23-gauge three-port pars plana vitrectomy (PPV) with internal limiting membrane (ILM) peeling assisted by membrane dual blue and tamponade with 16% hexafluoroethane (C2F6) gas. One month after surgery, OCT (RE) confirmed the successful closure of the macular hole with severe disruption of the ellipsoid zone (Fig. c). Due to posterior subcapsular cataract development in the RE, the patient subsequently underwent successful uncomplicated cataract surgery with phacoemulsification and intraocular lens implantation 6 months after PPV. At final follow-up, 11 months after PPV, the BCVA remained stable at 2.5/10 (RE) while the macular hole remained closed and a small part of the ellipsoid zone could be seen (Fig. e).
The scotopic full-field electroretinogram (ffERG) was non-recordable in either of the eyes indicating mostly loss of rod system sensitivity (Fig. a, b). The responses obtained from the photopic ERG, which entirely assesses cone function, were reduced and delayed in both eyes (Fig. c, d). These findings are in keeping with rod-cone dystrophy []. Due to the co-existence of severe bilateral sensorineural hearing impairment (data not shown), a clinical diagnosis of Usher syndrome was made.
After written informed consent had been obtained from the patient and family members (approved by the Bioethics Committee of the School of Medicine, Aristotle University of Thessaloniki), saliva samples were obtained from the patient and family members for subsequent genomic DNA extraction (Oragene DNA OG-500 saliva kits, DNA Genotek). Genetic testing following whole exome sequencing (WES) analyses revealed that the patient carried two compound heterozygous pathogenic variants in the USH2A gene (OMIM 608400, NM_007123.5) and in particular: a nonsense mutation, c.100C>T, p.(Arg34Ter), and a synonymous change c.949C>A, p.(Arg317=), which has been shown to lead to a new splice site []. The two variants, classified as pathogenic in ClinVar by multiple submitters, segregated with the phenotype in the patient’s family (Fig. ). Genetic counselling was given to the patient and family members.


A 27-year-old male, interior designer, presented to our clinic reporting a 4-month history of chronic pain within his left fourth toe that had been resistant to conservative management. He described his pain as sharp and continuous, and he was no longer able to wear closed shoes nor participate in his weekly indoor soccer games.
His medical history included type I diabetes mellitus controlled with a NovoRapid Flexpen® (insulin aspart) 100 U/mL (1 U/10 g administered by an insulin pump). His most recent HbA1c was 6.5%. He also experienced clinical depression treated with Zoloft® (sertraline hydrochloride), 50 mg each morning. He reported no allergies or sensitivities.
Physical examination identified moderate oedema affecting the left fourth toe in toto. Tenderness was elicited with palpation of the whole length of this digit and to a lesser extent the metatarsal head. This digit remained in normal alignment.
Diagnostic ultrasonography showed a small amount of soft tissue oedema within the digit. No evidence of a ganglionic cyst, tenosynovitis or bursitis was seen. X-rays exhibited a lucent lesion within the proximal phalanx measuring 15 mm × 7 mm (). Magnetic resonance imaging (MRI) () showed an expansile lesion breaching the inferior and lateral cortical bone. No soft tissue mass or definite stigmata suggestive of a chondrosarcoma was observed.


A 77-year-old Japanese woman diagnosed with Crohn’s disease of the small intestine had achieved remission with budesonide and maintained remission with mesalazine and enteral feeding. Six months after the initiation of therapy, she presented with fever, systemic punctate erythema, polyarthralgia, and bilateral coxalgia, with no relapse of gastrointestinal symptoms. Lower gastrointestinal endoscopy revealed that the lower ileal and colonic lesions were in remission. She was admitted to our hospital owing to difficulties in moving her body due to hip pain. On admission, she developed systemic punctate erythema with desquamation, polyarthralgia without redness and swelling, and hyperemia without ocular discharge on both bulbar conjunctiva (). The white blood cell count was 2510/μL and the serum C-reactive protein (CRP) level was 13.4 mg/dL. Short tau inversion recovery (STIR)-magnetic resonance imaging (MRI) of the sacroiliac joints revealed hyperintense foci in the bilateral sacroiliac joints, especially on the right side (). She was diagnosed with sacroiliitis, peripheral arthritis, skin rash, and scleritis associated with Crohn’s disease; treatment was initiated with NSAIDs and a topical preparation. However, owing to the poor response of bilateral coxalgia to this treatment, the patient was started on ustekinumab therapy. The Ankylosing Spondylitis Disease Activity Score (ASDAS)-CRP and Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) at the start of ustekinumab therapy were 4.3 and 7.0, respectively. Shortly thereafter, arthritis, skin eruptions, and scleritis resolved (), and the hip pain gradually improved. Two months after the start of ustekinumab therapy, STIR-MRI of the sacroiliac joints showed improvement in the white matter hyperintensities (), the ASDAS-CRP and BASDAI were significantly improved to 0.98 and 1.4, respectively, and the serum CRP level decreased to 1.39 mg/dL (). The patient has continued to receive ustekinumab every 8 weeks, with no relapse of any of the gastrointestinal, skin, and joint symptoms, including coxalgia. The HLA type determination performed later was positive for B52 and B54, but negative for B27.


A 48-year-old female, who recently returned from a holiday in Croatia, reported a 2-day history of coryzal symptoms and 1-day history of palpitations, dizziness and chest pain. Her past medical history included asthma and psoriasis. On presentation to the emergency department, she collapsed and was noted to be in polymorphic ventricular tachycardia, which developed into pulseless electrical activity (PEA) arrest requiring adrenaline and amiodarone, and three cycles of cardiopulmonary resuscitation (CPR). She was intubated and ventilated during the resuscitation.


A 61-year-old African American female with a history of end-stage renal disease due to adult polycystic kidney disease, s/p living-related kidney transplant in 2004 at an outside hospital, presented to the emergency room with fever for 5 days. She also had diarrhea and nausea, which had resolved by the time of presentation. The patient had nausea and diarrhea for 5 days. The diarrhea was nonbloody and watery, occurring about 4 to 5 times daily. The patient attended a funeral 10 days before her admission. She denied any history of cough, shortness of breath, chest pain, abdominal pain, vomiting, fatigue, myalgia, or headache. Symptoms started after attending a funeral in the city where several people had tested positive for COVID-19. Past medical history includes hypertension, gout, and histoplasmosis. Her home medications include tacrolimus 0.5 mg twice a day, mycophenolate mofetil (MMF) 250 mg twice a day, prednisone 5 mg daily, itraconazole 100 mg twice a day, allopurinol 100 mg daily, labetalol 100 mg twice a day, and hydralazine 100 mg 3 times a day.
On presentation, the patient was febrile with 103 °F, pulse rate 87 beats per minute, blood pressure 151/85 mm Hg, respiratory rate 24 breaths per minute, and oxygen saturation 92% to 97% on room air. Physical examination revealed that the patient was in respiratory distress with decreased breath sounds on the left side of the chest. The rest of the physical examination was unremarkable. The laboratory findings are summarized in . The patient’s baseline creatinine was 1.4 g/dL in the laboratory tests done in 2019. The urinalysis revealed 2+ protein, which was new, negative for blood and red blood cells. The chest X-ray revealed mild mid to lower lung infiltrate bilaterally, more significant on the left (). The computed tomography (CT) scan of the chest with contrast revealed few scattered rounded ground glass and consolidative opacities seen within the lingula of the left lung and middle lobe right lung and segmental lung consolidation seen within the dependent portion of both lower lobes ().
The patient was started on treatment with ceftriaxone and azithromycin for possible community-acquired pneumonia. The patient was started on intravenous fluids and antipyretics for fever. The patient’s MMF was held, tacrolimus and prednisone were continued. The COVID-19 testing done using polymerase chain reaction at the time of admission came positive. The patient needed only 2 to -3 L of oxygen for the first 2 days and no oxygen requirements after that. Her fever resolved. The patient was discharged after 5 days from the hospital. MMF was restarted and continued the same immunosuppressive regimen, before the hospital admission. The patient continues to do well after 2 weeks of discharge from the hospital.


A 73-year-old male presented to the emergency department with worsening intermittent diffuse abdominal pain and 40-pound weight loss over the past 3 months associated with increased flatulence, early satiety, and frequent explosive nonbloody diarrhea. The patient described the pain as cramping that typically worsened in severity at night. He denied fever/chills, dyspnea, cough, epistaxis, hematemesis, hemoptysis, hematochezia, and melena. The patient was a long-time science instructor at a local community college prior to transitioning to the Swedish import/export business with no significant exposure history, including asbestos. He had a prior history of right middle cerebral artery stroke and was a former smoker with a 60-pack-year smoking history.
Initial testing revealed normal electrolytes and liver function panel, elevated lactic acid (3.1 mmol/L), leukocytosis (white blood cell 12,100/mm3), and anemia (hematocrit 40.8%). Abdominal CT with oral and intravenous (IV) contrast revealed a homogenous retroperitoneal mass measuring 7.9 × 5.8 × 4.9 cm with displacement of the gastroesophageal junction and lesser curvature of stomach as well as a soft tissue mass involving the terminal ileum measuring 4.6 × 3.0 × 4.1 cm; both were concerning for neoplasm. Additional findings included diffuse lymphadenopathy in the retroperitoneum likely signifying metastatic disease. No evidence of obstruction was noted (see ).
Esophagogastroduodenoscopy confirmed concerns for extrinsic compression of gastroesophageal junction and fundus of stomach suggesting an underlying mass; no endoluminal gastric mass/lesions were appreciated (see ). Colonoscopy was attempted but scope was not able to advance past the sigmoid colon safely due to colonic stricture.
Computed tomography chest with IV contrast identified moderate diffuse emphysema, a right upper lung lobe spiculated mass measuring 4.5 cm, and indeterminate small left upper and lower lobe nodules (see ). Diffuse retrocrural and gastroesophageal lymphadenopathy was noted in addition to the retroperitoneal lymphadenopathy.
Given the concern for lymphoma versus alternative neoplastic disease, CT-guided biopsy was performed on the retroperitoneal abdominal mass and an intramuscular paraspinal mass. Hematoxylin and eosin staining revealed pleomorphic malignancy nuclei containing a vesicular chromatin pattern and prominent nucleoli (see ). Cells were predominantly polygonal with occasional spindling noted. IHC staining was positive for CK Oscar, cytokeratin 7, GATA3, calretinin, EMA, and CK5/6. Staining was negative for PAX8, TTE1, p40, p63, D2-40, WT-1, MOC31, CD34, MARI, CD45, dog 1, desmin, CD117, Sox10, actin, CA-9, and myogen.
These findings were suggestive of epithelioid MM with metastasis to the terminal ileum and lung; a diagnosis that was confirmed by Mayo Clinic Laboratories, noting this is a particularly atypical presentation of mesothelioma.
The patient opted for treatment of stage IV mesothelioma with plans to pursue a 21-day cycle of cisplatin, pemetrexed, and bevacizumab for up to 6 cycles. However, he presented to the emergency department 2 days after starting the second cycle with severe abdominal pain of 12 hours duration. He was found to have a moderate pneumoperitoneum with bowel perforation, worsening intra-abdominal mesothelioma, and several small bilateral acute renal parenchymal infarcts. The patient and his family opted for comfort care and he passed the following day.


A 7-year-old boy accompanied by his parents was referred to the department of endodontics at our institution by a private dentist, who had initially cared for the patient. As reported, the patient's right upper central incisor was avulsed while playing with his sister in a hammock. After communicating with the patient's father, who was at work near the family's residence, the boy was taken to the private dental office within 20 minutes. The avulsed tooth was kept dry. The dentist performed immediate replantation with the following protocol: 1) local anesthesia (Mepiadre 2%, DFL, Taquara, RJ, Brazil); 2) irrigation of the socket and avulsed tooth with saline; 3) tooth replantation; 4) semi-rigid orthodontic retention with photopolymerizable composite resin; 5) antibiotic prescription (amoxicillin; 250 mg, 5 mL every 8 hours for 7 days); 6) an analgesic (Alivium [ibuprofen]; 20 drops every 8 hours); 7) a tetanus vaccination assessment; and 8) resting orientation. The total extra-alveolar time between tooth avulsion and replantation was less than 30 minutes. The dentist responsible for the care referred the patient to an endodontic specialist.
After 3 days, the endodontist noted that the tooth was positioned with the incisal edge level inconsistent with the adjacent teeth; endodontic tests and radiographic examinations were performed, and a positive response to the cold test and incomplete rhizogenesis in tooth 11 were verified (). After initial analysis and verification that there were no other complications such as pain, pulp necrosis, and root fracture, it was decided to remove the semi-rigid splint after 15 days and to continue clinical and radiographic follow-up.
During the second session, a sinus tract originating in the root apex of tooth 11 was found (); therefore, we opted for pulp revascularization. The following sequence of clinical steps was performed: 1) removal of the semi-rigid containment; 2) local anesthesia; 3) absolute isolation; 4) coronal opening; 5) disinfection of the conduit with 2.5% sodium hypochlorite (Asfer Indústria Química LTDA, São Caetano do Sul, SP, Brazil); 6) irrigation with 17% ethylenediaminetetraacetic acid solution (Biodynamics, Lobato, PR, Brazil) for 3 minutes, 7) root canal filling with calcium hydroxide (Biodynamics), and 8) double sealing with glass ionomer cement (Maxxion R, FGM, Joinville, SC, Brazil). A return visit was scheduled for 21 days later.
At the follow-up session, the patient's father reported that the cavity seal had been accidentally removed; therefore, a new root decontamination session and application of intracanal medication were carried out as described above. After 2 weeks, blood clot formation was planned, but the patient still had a fistula. Therefore, we decided to perform further decontamination and to determine the actual tooth length (). After 14 days, when the patient returned without the fistula and with adequate coronal sealing, it was decided to attempt blood clot formation. The procedure included local anesthesia (Mepisv 3%, DFL) and promotion of bleeding inside the root canal by vigorous stimulation with 2-mm-caliber instruments longer than the actual tooth length (24 mm). Mild bleeding was detected inside the root canal; hence, the pulp chamber was cleaned, followed by root decontamination. Then, the root canal was filled with calcium hydroxide paste and a double seal was applied. Tactile detection of apical barrier formation was performed during this procedure.
Thirty days later, at the return visit, periapical radiography was performed to verify the presence of mineralized tissue in the root (). Formation of an apical mineralized barrier was observed. Therefore, we chose to apply stimulation to the root canal using mechanical instrumentation again. Subsequently, we apply calcium hydroxide to the root canal to stimulate the deposition of mineralized tissue (apexification). After 2 monthly changes, a radiographic examination verified the beginning of mineralized tissue formation between the cervical and middle third of the root canal (). In addition, minor intracanal bleeding was found during removal of the intracanal medication. Thus, we decided to renew the intracanal medication and perform clinical and radiographic analysis at a later session. At the follow-up session, an increase in the thickness of the apical barrier and between the cervical and middle third of the root canal () was detected. After this discovery, we opted to obturate of the portion of the root that was accessible via the canal.
Root obturation was performed 45 days after the last session by introducing Endosequence BC bioceramic cement (Brasseler, Savannah, GA, USA) without gutta-percha cones. On confirmatory radiography (), an absence of material in the mesial region was noted, and even after successive attempts, it was impossible to fill (). At subsequent consultations, restoration was performed with light-cured composite resin (Z250, 3M, Maple Wood, MN, USA), and further follow-up was scheduled.
To summarize, the treatment protocol used in this case was revascularization of the pulp of the replanted tooth. The technique was modified to include successive changes of intracanal medication before the use of biomaterial to control root infection, and the cervical third of the root was filled with a bioceramic material; in sequence, coronal restoration and clinical follow-up were performed.
At the 3-month follow-up after obturation, deposition of mineralized tissue beyond the initially formed apical barrier was noted (). Therefore, we chose to perform cone-beam computed tomography scans for a more detailed analysis of the root portion. Tomographic examinations over subsequent years showed root formation with an increase in thickness and length and absence of root resorption (). These data, combined with the absence of clinical symptoms and adequate coronal rehabilitation, demonstrated the success of the treatment.


A 3-year-old female child was transferred from peripheral hospital with abdominal aortic embolization of PDA device (Amplatzer ductal occluder, AGA Medical Corporation, Minnesota, USA) after failed percutaneous retrieval attempts.
Child was shifted to hybrid operating theatre and device retrieval was attempted under general anesthesia and endotracheal intubation with full heparinization (4 mg/kg). Retrieval attempts (by engaging the retention screw and snaring) failed due to the orientation of the device and also the device was snuggly fitting into the abdominal aorta []. Hence, surgical retrieval was planned. The option was device retrieval from the abdominal aorta through laparotomy and re-device or surgical PDA ligation through thoracotomy. To prevent laparotomy and its complications, hybrid approach was planned (the idea was to disengage the device from abdominal aorta and mobilize it to proximal thoracic aorta and retain it there till the aorta was looped via thoracotomy).
The ductus was crossed from the venous (antegrade) route and snared from the femoral artery to form an arteriovenous loop. Over this loop, a 12 mm TyShak® balloon (B. Braun Interventional Systems Inc. Bethlehem, PA, USA) was passed from the venous side. The inflated balloon was used to disengage the device from the abdominal aorta and mobilize it to descending thoracic aorta (DTA). The device re-embolization was prevented by keeping a partially inflated balloon (to allow distal blood flow) in the downstream of DTA through femoral artery guide wire (retrograde) [].
Then, a left thoracotomy was performed. Left subclavian artery, aortic isthmus, DTA, and PDA were dissected and looped using rubber slings. The DTA sling was partially snared to prevent device embolization and the TyShak balloon was deflated and withdrawn. PDA was doubly ligated using silk sutures. Then, the aorta was clamped distal to the subclavian and DTA, and device was retrieved through a small aortotomy, which was closed in 2 layers using 6-0 poly propylene sutures after de-airing. Heparin was reversed with protamine. Hemostasis confirmed and chest closed in anatomic layers with a single pleural drain in situ. The patient was shifted to intensive care unit and was extubated 6 h later and discharged on postoperative day 5. Bilateral femoral arteries and veins were patent at the time of discharge.
Hospital ethics committee waived of the clearance as parental consent was obtained.


A 30-year-old male patient known to have epilepsy was referred to the neurology clinic at our tertiary facility for evaluation and control of recurrent breakthrough seizures. He was born to nonconsanguineous parents, with an uneventful delivery and normal early development. He was of average intelligence and had completed 12 years of school education. Family history was negative for epilepsy, neurological or skin conditions. His epilepsy started at age 16 and has previously been controlled with sodium valproate 500 mg twice daily, with infrequent generalized tonic-clonic seizures over the last few years.
Neurological examination was normal. Electroencephalography “EEG” was unrevealing. Brain CT revealed a hypoattenuating lesion at the left parasagittal parietal area, with sulcal effacement and mass effect. It also revealed gyriform calcifications in the left fronto-temporo-parietal region, with bilateral cerebral atrophy and dilatation of both lateral ventricles which was significantly more prominent on the left side (). Incidentally, bone cysts were noted in the left mandibular condyle extending down to the ramus. Brain MRI with contrast () revealed a dura-based mass in the left parasagittal posterior parietal region, with enhancement and extensive surrounding vasogenic edema. It also confirmed the left gyriform calcifications, parenchymal atrophy and ventricular dilatation noted on brain CT. No vascular anomalies were noted in any of the images. The patient was referred to neurosurgery and the intracranial mass was excised (). Cytopathology and immunostaining revealed the mass to be due to fibrous meningioma, WHO grade I.
Also, during evaluation, it was noted that the patient had several left-sided skin lesions over his face (), scalp, posterior neck, abdomen, back and the left inguinal area. He also had multiple subcutaneous lipomas of different sizes, which were also restricted to the left side of his body. One of these lipomas was in the left frontotemporal area (). The skin lesions and lumps have been present for “as long as he could remember”. The abundance and diversity of lateralized Central Nervous System (CNS) and skin manifestations prompted a thorough evaluation for a neurocutaneous syndrome, and based upon a review of literature, ECCL was suspected. No intraspinal lipomas were revealed on whole-spine MRI. Biopsies from the skin lesions revealed them to be due to nevus sebaceous of Jadassohn, excluding nevus psiloliparis. There was no nonscarring alopecia, skin tags or aplasia. Ophthalmological evaluation was normal except for left melanosis sclerae. No evidence of aortic coarctation was discerned on clinical examination, chest x-ray or Transthoracic Echo. A comprehensive bone survey looking for cysts other than the ones detected in the left mandible was not performed, but none were detected upon revision of the available previous radiographs that were taken for different reasons.
During follow up, the patient suffered a left mandibular condylar fracture which was precipitated by a minor trauma and which was later complicated by osteomyelitis. Two years after meningioma excision, the patient’s epilepsy is again well controlled with medical treatment. This case demonstrated involvement of three systems (skin, central nervous system, and other), with major criteria in the latter two. Based on these findings, and the exclusion of alternative diagnoses, it meets Moog’s criteria for a definite case of ECCL.


A 71-year-old female with a history of type 2 diabetes mellitus, hypothyroidism, interstitial cystitis, and osteoarthritis presented with clinical stage T1N0M0 ER+/PR–/HER2– invasive ductal carcinoma of the lower outer quadrant of the left breast, for which she underwent left segmental mastectomy and sentinel lymph node biopsy. Pathology demonstrated a 14-mm grade 2 invasive ductal carcinoma with ductal carcinoma in situ, positive lymphovascular invasion, and ductal carcinoma in situ present at the margins. One of 3 sentinel lymph nodes was positive with 8-mm macrometastasis and 4.5-mm extracapsular extension that was not present on gross examination. She then underwent re-excision of the breast with negative final margins and completion axillary lymph node dissection, which identified 13 lymph nodes, all negative for carcinoma. Oncotype testing was ordered, but processing issues occurred, resulting in a delay. The decision was thus made to proceed with adjuvant radiotherapy within 8 weeks of the surgery date.
RT was delivered to the whole left breast using active breathing control to a dose of 42.4 Gy in 16 fractions with opposed photon tangents with a field-in-field technique to minimize dose heterogeneity using 6- and 8-MV photons delivered by a linear accelerator. The patient received a sequential tumor bed boost without active breathing control to a dose of 10 Gy in 4 fractions using a 3-dimensional conformal technique with 6-MV photons. The ipsilateral lung V4Gy, V16Gy, and V20Gy were 27, 14, and 12%, respectively, and the mean heart dose was 1.04 Gy. During treatment, the patient complained of intermittent burning and mild discomfort in the breast, alleviated by daily acetaminophen. Mild skin erythema was noted, and the patient applied Miaderm twice daily to her breast during treatment. She completed RT without interruptions; no desquamation was observed at the last on-treatment visit.
After completing radiation, the patient's OncotypeDX returned with a score of 31, for which she was advised to pursue adjuvant chemotherapy. Docetaxel and cyclophosphamide were initiated 3 weeks after RT completion and planned for 4 cycles. Prior to initiation of chemotherapy, her skin was intact with erythema limited to the axilla. She then presented with left breast tenderness, erythema, and warmth with no fevers 1 week after starting chemotherapy and 4 weeks after completing RT (Fig. ). At the time of physical examination, the differential diagnosis included cellulitis versus RRD, and a trial of cephalexin for 1 week was prescribed. After no improvement, she was prescribed oral prednisone at 20 mg daily for 7 days and silver sulfadiazine for presumed RRD. The patient returned to the clinic 1 week after starting prednisone therapy and reported marked improvement in her skin appearance (Fig. )C. She was premedicated with 20 mg of prednisone for cycle 2 of chemotherapy. However, she developed less severe RRD after her second cycle of chemotherapy, so prednisone was increased to 30 mg, followed by a brief taper for the third and fourth chemotherapy cycles. The patient tolerated the remaining chemotherapy cycles well and successfully completed chemotherapy.
When the patient began letrozole 3 weeks after having completed all chemotherapy, she reported breast warmth similar to what she had experienced before developing RRD, although the clinical examination was unremarkable (Fig. ). After discussion with a medical oncologist, letrozole was held for 2–3 weeks. As there were no changes to the breast during this time, letrozole was reinitiated. Her breast warmth and heaviness were attributed to breast lymphedema, and she continued physical therapy management.
At her 6-month post-radiation treatment follow-up, she reported having had a dry cough for 2 weeks without fever; a chest X-ray revealed consolidation in the left middle lung concerning for pneumonia. Of note, she had a computed tomography (CT) chest scan performed 1 month prior to radiation as part of a research study, which had shown postradiation changes (Fig. ). For her symptoms and abnormal X-ray, she was prescribed azithromycin and benzonatate. However, she then became febrile to 101.5°F and presented to the emergency department, where a CT angiogram of her chest showed evidence of left upper- and left lower-lobe infiltrates (Fig. ). She was admitted for intravenous antibiotics but showed no improvement and developed increased shortness of breath. Bronchoscopy with bronchoalveolar lavage revealed elevated lymphocytes (73%), monocytes (2%), and eosinophils (12%) on the cell count differential, and cultures showed only normal respiratory flora. The patient was thus initiated on prednisone at 60 mg daily for presumed radiation pneumonitis; she reported an improvement in symptoms within 1 day. A chest CT completed around 1.5 months later demonstrated improvement in the consolidation, and the patient had returned to her baseline respiratory function clinically and on pulmonary function tests (Fig. ). She completed prednisone tapering approximately 1 month later.
The patient was admitted again to the hospital 1.5 months after completing the steroid taper (9.5 months after completing RT) due to fatigue and shortness of breath. A chest CT showed bilateral multifocal rounded consolidations and airspace disease concerning for organizing pneumonia (Fig. ). Bronchoscopy with bronchoalveolar lavage showed 2% lymphocytes, 81% monocytes, and 3% eosinophils. Cultures revealed only normal respiratory flora. A biopsy of the right lung revealed organizing pneumonia (Fig. ) with stains negative for fungal organisms or pneumocystis. She briefly required supplemental oxygen after a bronchoscopy but was discharged without oxygen requirement. She was placed on 60 mg prednisone following the biopsy results and prophylactic sulfamethoxazole/trimethoprim with pulmonological follow-up. Two months later, she reported no shortness of breath and only occasional cough on 30 mg prednisone, with a chest CT demonstrating resolution of the prior airspace consolidation (Fig. ). She did report side effects from the steroids, including insomnia, a 16-lb weight gain over the previous 2 months, and elevated blood glucose levels. She was advised to complete a steroid taper over the next 2 months with further follow-up.


In the 18th week of gestation, routine ultrasound examination of a 35-year-old primigravida revealed a fetal cystic intra-abdominal mass occupying the entire abdominal cavity. A prenatal magnetic resonance imaging (MRI), performed in the 29th gestational week, showed a lymphatic cystic malformation measuring 12 × 7 × 9 cm (). In the 33rd gestational week, intestinal hypoperfusion due to compression by the malformation was suspected during an ultrasound examination. Therefore, a caesarean section was performed without complications.
After birth, the male baby presented in good general condition with stable vital signs. The abdomen was distended but not painful on palpation. Postnatal ultrasound confirmed the lesion. To assess the exact extend, MRI was performed showing a macrocystic LM occupying the entire abdomen () without ascites. However, the exact origin of the lesion and the amount of extension into the mesenteric root could not be described.
On the 13th day of life, the boy suddenly developed massive abdominal distension and compartment syndrome requiring tracheal intubation because of respiratory distress. Hemoglobin dropped from 15.7 to 9.6 g/dl within 3 hours and lactate was elevated with 8.8 mmol/l. Emergency laparotomy was performed for decompression and demonstrated that the cystic mass occupied the ileocecal mesentery. Two major cysts were hemorrhagic explaining the abdominal compartment syndrome (). An extended ileocecal resection allowed the complete removal of the LM. Subsequently, a termino-terminal ileocolic anastomosis was created ().
Histological examination confirmed the diagnosis of a cystic LM with acute hemorrhage.
The postoperative course was uneventful. Abdominal ultrasounds were performed 1, 3, 6, 12 and 18 months postoperatively and were without evidence of recurrence.


A 62-year-old male with hypertension and hyperlipidaemia presented to an urgent care clinic with 7 days of dizziness, fatigue, nausea, and vomiting. He was transported to the emergency department, evaluated, and discharged home. Approximately 4 days later, emergency medical services were called after the patient experienced syncope. Electrocardiogram (ECG) obtained en route reported anterior ST-segment elevations. Before arrival at the hospital he developed ventricular fibrillation. Cardiac defibrillation was successful and endotracheal intubation was performed. Upon arrival at the emergency department, ventricular fibrillation recurred, and resuscitative efforts restored sinus rhythm and spontaneous circulation. Following return of spontaneous circulation, exam was notable for heart rate of 77 b.p.m., a regular rhythm, and hypotension (86 mmHg/48 mmHg). There were no appreciable murmurs. There was no lower extremity oedema. The extremities were cool to touch. On pulmonary exam, he required mechanical ventilation with a rate of 28, tidal volume 500 mL, FiO2 100%, and positive end-expiratory pressure of 8 cmH2O. Breath sounds were present bilaterally. The subsequent ECG showed wide complex rhythm consistent with slow ventricular tachycardia with right bundle branch block morphology and left axis deviation. ().
Given the report of ST elevations prior to ventricular fibrillation, along with the ECG at presentation, there was high suspicion for acute coronary syndrome. He received aspirin, ticagrelor, and heparin for a presumed ST-segment elevation myocardial infarction. Epinephrine infusion was started due to bradycardia with hypotension. He was emergently taken to the catheterization lab, but coronary angiography did not reveal coronary stenosis. Right heart catheterization revealed elevated right-sided filling pressures with a right atrial pressure of 22 mmHg, pulmonary artery pressure 61/28 (39) mmHg, pulmonary capillary wedge pressure 15 mmHg, and Fick cardiac index 2.6 L/min/m2. Due to the unclear nature of his presentation, point-of-care echocardiogram was performed, and demonstrated a dilated right ventricle with severely reduced function. Based on point-of-care echocardiogram findings, there was concern for pulmonary embolism. Immediate pulmonary angiography was performed and revealed large, bilateral pulmonary emboli (, Video ). EkoSonic™ endovascular thrombolysis catheters were advanced into both main pulmonary arteries and 5 mg of tissue plasminogen activator was delivered through each catheter, followed by 2 mg/catheter/h for 2 h, then 1 mg/catheter/h for 16 h. Infusion was guided by fibrinogen monitoring as per institutional protocol.
Upper and lower extremity Doppler ultrasounds were obtained but showed no evidence of venous thrombosis. Formal transthoracic echocardiogram confirmed depressed right ventricular function (, Video ). Computed tomography (CT) of the chest showed bilateral peripheral ground-glass opacities with wedge-shaped opacities in the right lung (). These were thought to represent pulmonary infarctions, but, given refractory hypotension requiring vasopressors, he was started on broad-spectrum antibiotics for pneumonia. A viral respiratory panel was negative, but tracheal aspirate culture was positive for methicillin-resistant Staphylococcus aureus. Following completion of catheter-directed thrombolysis, repeat ECG showed sinus rhythm with first-degree atrioventricular (AV) block, left axis deviation, incomplete right bundle branch block, and prolonged QTc interval (498 ms) (). Over the following 4 days, he developed anaemia, and CT of the chest, abdomen, and pelvis showed a mediastinal haematoma and persistent ground-glass opacities (). Given his radiographic findings, and growing prevalence of COVID-19, he was tested for SARS-CoV-2 and found to be positive. He was transferred to a COVID-19-dedicated intensive care unit where he was placed under enhanced contact precautions and received supportive care. He was extubated 4 days later. Following extubation, he did well. He was admitted to an inpatient rehabilitation facility and discharged home on lifelong apixaban 5 mg twice daily. At 1 month follow-up, he described mild exertional dyspnoea that was improving. Transthoracic echocardiogram at that visit noted improvement of right ventricular dilation and systolic function (, Video ).


A 54-year-old man complained of aggravating abdominal pain 15 h after endoscopic resection of a colonic LST.
The patient was referred to our hospital for endoscopic resection of an LST measuring 30 mm that was diagnosed as an adenoma on pathology in the medial side of the colonic hepatic curvature. The patient was placed on a low-fiber diet on the day prior to endoscopic surgery. Adequate bowel preparation (BBPS: 3 points) was achieved using a spilt regimen of 4 L of polyethylene glycol solution and simethicone. During the procedure, our experienced team performed ESD for this lesion at first. When damage to the muscular layer caused by poor scope operability was observed, we changed into EPMR to quickly finish the operation. Prophylactic closure with clips was performed at the damage site. The tumor was removed without residue, and the wound was treated quite well without obvious perforation or hemorrhage. There was no abdominal pain after resection, and the patient was in a good condition. Routine antibiotics and intravenous nutrition were used in consideration of the large mucosal defect. Fifteen hours after endoscopic resection, the patient passed gas and then suddenly experienced severe abdominal pain that aggravated progressively over time without any alleviation.
The patient had a history of diabetes for 10 years without a regular oral hypoglycemic treatment. He had no history of other chronic diseases like cardiovascular disease and hypertension, and no history of trauma, surgery, or blood transfusion. He had no history of food and drug allergy.
The patient occasionally drank alcohol and denied a history of smoking. He had no family history of intestinal tumors.
Physical examination showed intolerable compression pain and rebound pain localized in the right abdomen, suggesting the presence of peritonitis.
Emergency blood analysis showed that neutrophil proportion increased slightly (79.9%) with normal white blood cell count, hemoglobin, and red blood cell count.
Emergency computed tomography (CT) revealed a small amount of pneumoperitoneum and substantial exudate (Figure ).


Rash, fever for 8 d, and dyspnea for 12 h.
The 42-year-old Chinese woman initially presented with fever and a small rash on her forehead and face. Over the next 2 d, she developed a diffuse, pustular, itchy rash over her trunk and legs. She also complained of a cough with white foam-like sputum, and chest tightness. Four days later, due to dyspnea, her mental status started to gradually deteriorate. She required supplemental oxygen. She then became more and more drowsy. Six days later, she received endotracheal intubation and ventilator support and was admitted to the intensive care unit. She was a housewife, 1.62 m in height with a body weight of 55.4 kg.
She had a history of lumbar disc herniation. She had taken traditional Chinese medicine for back pain 1 d before the onset of fever and rash.
She had no other past medical history, and no family history of similar diseases or psoriasis.
Physical examination showed diffuse, pustular rashes over her trunk and extremities (Figure ).
Laboratory studies showed a white cell count of 15.3 × 109/L, neutrophils of 87.3%, C-reactive protein of 208.00 mg/L, and procalcitonin of 0.18 ng/mL. Blood gas analysis showed pH 7.35, PaO2 62.1 mmHg, PaCO2 58.3 mmHg, and HCO3- 31 mmol/L. A large number of white cells were seen in the pustular fluid smear. Pustular fluid culture was negative. Anti-nuclear antibody, anti-neutrophil cytoplasm antibody, anti-mitochondrial antibody, TB-GeneXpert, and T-spot were within normal limits. All blood and pustular fluid cultures were negative.
Lung computed tomography showed that the airway space was occupied and tracheoscopy was recommended. Tracheoscopy revealed an unidentified protrusion in the main bronchus (Figure ).
Skin and airway mucosa biopsies showed subepidermal bullous formation containing scattered neutrophils and eosinophils with occasional subcorneal neutrophilic pustules, which suggested the diagnosis of AGEP (Figure ).


A 42-year-old anemic female presented to the clinic, complaining of a recurrent obturator hernia for the past 10 years. It was a large, painless, progressive swelling in the perineum, involving the labial fold. There were no bowel obstruction symptoms. She was previously operated for the hernia three times in different centers. On examination, there was a large swelling extending from the right medial gluteal area to the right labial fold measuring 20 × 15 cm. It was nontender and had a firm consistency, with no overlying skin changes (). Laboratory investigations were within normal limits. Computed tomography (CT) scans of the abdomen and pelvis showed a massive, multicompartmental, multilobulated soft-tissue lesion in the lower pelvis and perineum, exerting a mild mass effect (). Perineal mass excision, possible mesh placement and stoma creation was planned. At exploratory laparotomy, the abdominal and pelvic cavities were examined. There was no evidence of a defect or hernia. The extraperitoneal space was explored and the obturator foramen was identified without any hernia. The procedure then shifted to excision of the mass. It was lobulated within a capsule and extending from the vulvar commissure to the anus. It was also involving part of the lateral vaginal wall, the lower part of the rectum and anal canal which needed to be excised (). The mass was directly subdermal at the vulvar commissure site, so part of the labia minora skin was excised with the mass. The vaginal wall was repaired by a urogynecologist. The rectum was repaired by the primary surgeon. A diversion end sigmoid colostomy was created. Wound closure was assisted by plastic surgery. No tissue or skin coverage was required. The histopathological report revealed an AA extending to the margins with ulcerated and reactive skin tissue. Immunostaining for estrogen and progesterone were both positive. The postoperative course was uneventful. At 8 months follow-up, abdomen and pelvis CT concluded that there was a minimal residual perineal mass without extension to the pelvis. Following discussion in the multidisciplinary tumor board, she was started on hormonal therapy after ~1 year of surgery with leuprolide 22.5 mg, intramuscular, every 3 months. Hormonal therapy was continued for a total of 15 months with frequent follow-up images which showed no change in the residual mass. She underwent colostomy reversal 3 years after and was doing well on regular follow-ups. The patient consented to the publication of this case report with images. All measures were taken to ensure her privacy and anonymity.


A 70-year-old male without significant medical history except for being a former smoker presented to our emergency service with the left shoulder pain for the past 2 weeks. The pain was located in the distal third of the clavicle, and there was no history of trauma.
On examination, there was a significant pain on palpation in the distal third of the clavicle and acromioclavicular joint. Limited active range of motion (abduction of 100°, anteflexion 90°, external rotation of 20°, and internal rotation up to L3), and abnormal rotator cuff strength (4 of 5).There was no vascular or neurologic deficit.
Standard two-dimensional radiographs were taken (), with an abnormal fracture of the lateral third of clavicle without clear traumatic history. Magnetic resonance imaging showed a fracture in the lateral third of clavicle accompanied by a soft tissue mass and bone edema ().
On suspicion of a pathological fracture, a positron emission tomography/computed tomography (PET-CT) scan was performed to further assess the lesion and as a staging study. It showed at the PET-CT appreciates metabolically active pulmonary mass in the left upper lobe, mediastinal and left hilar adenopathy, hepatic lesions, and a bone lesion at the distal end of the left clavicle (), left sternoclavicular joint, left the transverse process of the 8th vertebra, and right transverse process of the 9th vertebra.
After having the staging studies, an ultrasound-guided biopsy was performed of the distal third clavicle lesion. The results of the biopsy came back compatible with a neuroendocrine carcinoma based on immunohisto chemical results (P63+, TTF1 -. CD56+ Ki67 80%). A transbronchial biopsy of the left upper lobe mass resulted in a small cell lung carcinoma.
The patient was diagnosed with Stage IV advanced stage lung carcinoma and died 6 months after having with the pathological clavicle fracture.


A three-year-old female with a history of complex congenital heart disease and previous cardiac surgeries was admitted to the pediatric intensive care unit (PICU) in shock that progressed to cardiopulmonary arrest requiring resuscitation
due to thrombosis in her right ventricle-to-pulmonary artery conduit. She subsequently demonstrated evidence of severe hypoxic-ischemic encephalopathy and acute kidney injury (AKI), of which the latter resolved.
The main reason for her subsequent PICU stay was for neurological rehabilitation and transition to home ventilation. She was receiving treatment for dystonia, heart failure, endocarditis, and pulmonary embolism. At the request of the parents, stem cell therapy for neurological dysfunction was investigated. As metformin has shown properties to recruit existing neural stem cells and enhance neural functions, a trial of therapy was initiated. The patient was placed on metformin (20 mg/kg) 17 days before the event, which was increased to 40 mg/kg (250 mg twice-daily) six days prior to the event.
On the day of the event, the patient acutely presented with a new wide complex bradycardia 70–80 beats per minute (bpm) from a baseline of 110–120 bpm and peaked T waves. The other vital signs were blood pressure = 90/40 mmHg, respiratory rate = 26 breaths per minute, temperature = 37.2 °C, and oxygen saturation = 87% on her baseline ventilation settings. She was poorly perfused with prolonged capillary refill time and lethargic. Immediate and subsequent venous blood sampling showed severe lactic acidosis and hyperkalemia [].
She was managed for hyperkalemia, and propranolol was discontinued. She became hypotensive, requiring dopamine and norepinephrine infusions, and higher ventilator support. The patient’s clinical status improved within three hours, and support therapies were weaned towards the baseline. Twelve hours after the initial event, a similar episode recurred with a rise in serum lactate and hyperkalemia, which was treated similarly and resolved. A detailed review of both events demonstrated that they occurred 1 to 2 hours after the administration of metformin. In addition, her creatinine levels had doubled five days prior to the event with a recent increment in metformin dose six days before.
At this point, metformin was discontinued. No further episodes were noted, and her clinical status returned to baseline [].


A 26-year-old male handworker with a history of surgically treated pigmented villonodular synovitis (PVNS) of the right knee 2 years ago presented in the orthopedics clinic with right knee hemarthrosis. On initial examination, his knee showed an inability of fuel weight-bearing, and the range of motion was limited to 30°. After a joint aspiration of 120 ml of blood, the patient was treated with NSAIDs, which led to a symptom relief for 15 days. However, during the following 5 months, he experienced six more episodes of hemarthrosis, two of which required arthrocentesis. Microorganism culture was negative, while the cytological examination showed hemorrhagic items and chronic inflammatory response. In a search for potential bleeding disorders, the patient underwent a series of blood tests including complete blood count, platelet aggregation test, and bleeding time test, which were all negative. A knee magnetic resonance imaging (MRI) was performed after the 2nd arthrocentesis, which showed an anterior cruciate ligament graft tension relaxation and arthritis.
As a next diagnostic step, an arthroscopy was decided, during which synovial biopsy and synovial fluid analysis was performed. The results of the synovial biopsy revealed chronic synovitis and hemochromatosis. The patient remained symptom free for 1 month but relapsed again with a sudden episode of hemarthrosis during walking. Despite the performance of a joint aspiration of 100 ml of blood and the intra-articular injection of betamethasone, the symptoms persisted. In this context, a vascular dysplasia was suspected, but MRI angiography was negative. A second arthroscopy revealed no specific cause. Osmic acid synovectomy of the knee was performed, but after a short period of symptom alleviation, knee hemarthrosis relapsed [].
After the failure of all the above-mentioned diagnostic and therapeutic approaches, a knee radiosynoviorthesis with the radioisotope yttrium-90 (90Y) as symptomatic treatment was suggested. The aim of the therapy was to diminish the hemorrhagic synovium. The patient had a preradiosynovectomy three-phase bone scintigraphy, which confirmed increased 99mTc-MDP uptake in the blood pool phase, as a sign of inflammation [].
The radiosynovectomy procedure was performed under strict aseptic conditions. After local lidocaine injection for analgesia, excess joint fluid was drained, and 5 mCi of 90Y was injected intra-articularly into the knee joint followed by the administration of 20 mg of triamcinolone acetonide to diminish the risk of an acute inflammatory response and to prevent radiopharmaceutical skin contamination from the removal of the needle. Immediately after 90Y application, static imaging of the ankle joint was performed to demonstrate the distribution of the radiopharmaceutical in the joint and exclude potential extra-articular leakage [].
The patient was in stillness for the next 48 h and was instructed to avoid strenuous physical activity during the 1st day after treatment. Gradually, he followed a program of mobilization and empowerment with improvement of his symptoms as early as 1 month after the first radiosynovectomy. However, due to residual joint effusion, the second radiosynovectomy was performed 6 months later, following the same procedure. Soon after application of the second synovectomy, the patient’s symptoms almost completely subsided, and he returned to his work. More than 4 years after of the radiosynovectomy, the patient remains symptom – free, functional and is working on a full-time schedule []. No side effects were reported with both radiosynovectomy procedures.


A 72-year-old woman was referred for evaluation of a 10-year history of chronic
intermittent pain in the right loin. Her numeric rating scale score for pain was 7 on a
scale of 0 to 10, where 0 indicates no pain and 10 indicates the worst pain, and was
exacerbated by walking. She had no specific history of trauma. Before visiting the pain
clinic, she had been examined by a gastroenterologist and a nephrologist. A left renal
cyst and a small hepatic cyst were found in a computed tomography (CT) scan of the abdomen
and kidneys. Nevertheless, the nephrologist could not identify the cause of her pain, and
she was referred to a pain clinic. Physical examination revealed tenderness throughout the
rib margin on the right side, and her symptoms were reproduced by the hooking maneuver. A
twelfth intercostal nerve block was performed under ultrasound guidance with triamcinolone
(10 mg) in 5 mL of 0.4% mepivacaine, and her pain was immediately and completely relieved.
One week later, the patient said that she was doing well without pain, and she was lost to
follow-up.


A 47-year-old woman was hospitalized for loin pain and consulted with the pain clinic
because the cause of the pain had not been found despite various tests. She had a 3-month
history of intermittent pain in the loin and both lower rib cages. The pain was sharp and
intermittent and was aggravated by walking. Her numeric rating scale score for pain was 8.
She had no history of trauma or injury. All radiological findings were normal, including
an abdominal CT scan, bone scan, and X-rays of the rib cage. Physical examination revealed
severe tenderness in the subcostal area that was reproduced by the hooking maneuver. Her
pain was completely relieved following twelfth intercostal nerve block under ultrasound
guidance using triamcinolone (10 mg) in 5 mL of 0.4% mepivacaine. She remained well
without pain for 1 week, at which time she was discharged. She was thereafter lost to
follow-up.
Neither patient developed complications after the intercostal nerve block. This study was
approved by our institutional review board (approval No. 2020-03-014). The board waived
the requirement for informed consent.


A 27-year-old male patient with no significant past medical history was admitted to the orthopedic emergency department of our hospital with a painful locked right knee. There was no history of either trauma or any unusual sports injury. He reported suffering from mild pain, which was worse with activity, and a decreased range of motion for two years but denied any swelling or locking of the knee during this period. Physical examination revealed massive swelling and tenderness along the joint line and increased pain with active and passive motion of the knee. Specific knee joint tests could not be performed due to excessive pain. There were no signs of inflammation (erythrocyte sedimentation rate, C-reactive protein, and total blood count tests were normal). Plain radiography of the knee joint was also normal. Magnetic resonance imaging (MRI) revealed massive swelling and a well-circumscribed lobulated intraarticular mass, 4x2x1.5 cm in size, originating from the distal one third of the patellar side of the ligamentum mucosum (Figure 1a-c). However, no meniscal, ligamentous, or cartilaginous pathology was observed. Due to the imaging findings, GCTTS or fibroma was our primary consideration.
The arthroscopic procedure was performed under general anesthesia through standard anteromedial and anterolateral portals. After entry through the anterolateral portal, synovial fluid samples were taken for culture and Giemsa staining, which were negative. Following the sampling, the area was evaluated and the mass was identified as mainly originating from one third of the patellar side of the ligamentum mucosum (Figure 2a). Then, after entering the anteromedial portal, the mass was removed by enlarging the anteromedial portal with the help of a punch (Figure 2b). Finally, the hypertrophic and inflamed synovial tissues were debrided with an arthroscopic shaver and radiofrequency ablation.
Macroscopically, the resected mass was 4x2x1.5 cm in size, with a smooth surface, lobulated contour, and brown color (Figure 2c). Histopathologically, the mass was composed of small and round mononuclear cells and osteoclast-like giant cells (Figure 3). The lesion was diagnosed as localized GCTTS. At two-year follow-up, the patient remained asymptomatic and MRI revealed no recurrence of the lesion (Figure 4a-c). A written informed written consent was obtained from the patient.


A 67-year-old female patient presented with one-month history of neck pain exacerbated by movement. On clinical examination, there was no motor weakness or sensory deficit. The patient was afebrile. On laboratory examination, the patient had normal hemoglobin levels, normal liver function test results, and normal results on uric acid, calcium, phosphorus, and urine analyses. Her erythrocyte sedimentation rate (ESR) was 15 mm/hour (normal, 0-10 mm/hour) and C-reactive protein (CRP) increased to 3 mg/dL (normal, 0-0.3 mg/dL). A written informed consent was obtained from the patient.
Magnetic resonance imaging (MRI) showed marrow edema within the dens, anterior arch of C1 and pathological contrast enhancement at periarticular soft tissue (Figure 1a-d). Computed tomography (CT) revealed periodontoid calcified deposits suggesting CPPD (Figure 1e). Magnetic resonance imaging also demonstrated bone marrow edema on C4-5 facet joint and perivertebral signal alteration adjacent to the facet joint (Figure 2). The patient was diagnosed as CPPD disease and started systemic antiinflammatory treatment (oral colchicine). At one-year follow-up, the neck pain gradually improved.


A 70-year-old Korean female was referred to our hospital because of massive hemoptysis and modified Medical Research Council (mMRC) grade 4 dyspnea on September 19, 2008. Three days before this admission, she had abdominal pain, fever, chills, myalgia, and nonproductive cough. She had a history of hypertension. She was a farmer and worked mainly in the fields. Upon physical examination, she had low blood pressure (80/60 mmHg), tachypnea (30 breaths/min), tachycardia (120 beats/min), inspiratory crackle on both lung fields, and conjunctival suffusion. Chest radiography revealed bilateral areas of diffuse infiltrative opacification. A computed tomography (CT) scan of the chest showed diffuse ground-glass opacities (GGO), interlobular septal thickening, and a moderate amount of pleural effusion in both lungs. Laboratory findings showed white blood cells of 9,980/µL (normal range, 4,000–8,000/µL), hemoglobin of 8.1 g/dL (normal range, 12–16 g/dL), platelets of 163 × 103/µL (normal range, 150–400 × 103/µL), and creatinine of 1.47 mg/dL (normal range, 0.6–1.4 mg/dL). Arterial blood gases revealed a pH of 7.406, pCO2 of 26 mmHg, and pO2 of 49 mmHg with FiO2 of 90%. The patient was immediately transferred to the intensive care unit and intubated due to acute respiratory failure. She was treated empirically with oral doxycycline and intravenous ceftriaxone. At 4 and 13 days after symptom onset, the antibody titers using passive hemagglutination assay (PHA) were 1:1,280 and 1:20,480, respectively. N-PCR for Leptospira spp. showed a positive result from urine 9 and 15 days after symptom onset, but N-PCR with the plasma and buffy coat 16 and 18 days after symptom onset was negative. Six days after treatment, the respiratory symptoms and hypoxemia had resolved, and extubation was performed. The patient was discharged home 18 days after symptom onset. At that time, N-PCR using a urine sample was positive.


A 73-year-old Korean female was admitted to the emergency room with a 6-day history of headache, myalgia, fever, and chills on August 9, 2007. She had no medical history. She reported that she had worked in the fields 10 days before the admission. She presented with fever (37.7°C). Laboratory results showed white blood cells of 13,480/µL, hemoglobin of 10.4 g/dL, and platelets of 96 × 103/µL. A lumbar puncture test was normal. The antibody titer using PHA was negative at admission. Supportive treatment was performed considering that the fever was due to a viral infection. However, the antibody titer 10 days after symptom onset was 1:5,120. At the same time, Leptospira spp. N-PCR with urine was positive, and also, N-PCR with plasma showed a positive result 11 days after symptom onset. Thereafter, treatment with doxycycline was started. She was discharged home after a 14-day course of doxycycline.


An 80-year-old Korean female, who had no previous medical history, was admitted with a headache on September 6, 2007. Six days before admission, she had myalgia, chills, fever, and a sore throat. Upon physical examination, she had a high-grade fever (38.6°C). The laboratory results showed white blood cells of 2,890/µL, hemoglobin of 13.2 g/dL, and platelets of 112 × 103/µL. Urinalysis showed 1+ protein, 4+ blood, with many red blood cells, and 0–1 white blood cells/high-power field. A lumbar puncture test was performed. The CSF showed a white blood cell count of 57/µL, neutrophils of 83%, lymphocytes of 13%, protein of 42 mg/dL, and glucose of 71.9 mg/dL. The antibody titer at admission was negative. N-PCR for Leptospira spp. with plasma showed a negative result, but N-PCR with urine and CSF was positive 8 days after symptom onset. The antibody titer was 1:10,240 12 days after symptom onset. She was discharged home with a 14-day course of doxycycline.


A 76-year-old Korean female was referred to our hospital because of an altered mental status on August 30, 2007. Four days before the admission, she presented with general weakness and myalgia. She had no medical history. Upon physical examination, she had stuporous mentation, low blood pressure (80/50 mmHg), tachypnea (28 breaths/min), tachycardia (108 beats/min), and an inspiratory crackle on both lung fields. The laboratory results showed white blood cells of 10,190/µL, hemoglobin of 9.8 g/dL, platelets of 83 × 103/µL, and creatinine of 2.31 mg/dL. Urinalysis showed 1+ protein, 5+ blood, with many red blood cells, and 5–9 white blood cells/high-power field. Chest radiography revealed bilateral areas of diffuse infiltrative opacification. After admission, the patient was intubated due to acute respiratory distress. A CT scan of the chest showed bilateral areas of ground-glass attenuation with superimposed interlobular septal thickening. The antibody titer was negative 5 days after symptom onset, but N-PCR with a plasma sample was positive 6 days after symptom onset. The antibody titer was 1:1,280 10 days after symptom onset. After 5 days of treatment with doxycycline, the patient was extubated. Shortly thereafter, she went into acute respiratory distress and died, although mechanical ventilation was applied.


The patient was a 36-year-old female with a history of morbid obesity, major depressive disorder, borderline personality disorder, posttraumatic stress disorder, migraine headaches, and back pain. She possessed the decisional capacity to provide informed consent. She was under the regular care of an internist and a psychiatrist. She had tried numerous weight loss strategies without sustained success. The patient underwent laparoscopic sleeve gastrectomy, after receiving nutrition counseling and psychological clearance for the procedure. An upper gastrointestinal series done prior to the procedure was normal. An x-ray esophagram showed interrupted primary peristalsis with mild stasis, with eventual clearing with secondary peristalsis. The study was otherwise unremarkable. The patient’s weight at the time of surgery was 269.6 pounds (body mass index (BMI) = 41.0). The patient underwent the sleeve gastrectomy with no complications. Following surgery, she experienced intermittent nausea and vomiting. She had difficulty with swallowing all of her medications, which were liquid, crushable tablets, or capsules whose contents could be emptied into liquid. Medications included promethazine suppositories, ondansetron rapidly dissolving tablet, hydrocodone/acetaminophen solution, acetaminophen-butalbital caffeine tablet, pantoprazole granules, suvorexant tablet, zolpidem tablet, gabapentin capsule, multivitamin tablet, lurasidone tablet, prazosin capsule, duloxetine capsule, trazodone tablet, fexofenadine tablet, propranolol tablet, dicyclomine capsule, sumatriptan injector, and lithium carbonate capsule (300 mg taken in the morning and 600 mg taken in the evening).
Prior to the sleeve gastrectomy, the lithium level had been 0.62 mmol/L. About 4 months after the surgery, the lithium level had increased to 1.47 mmol/L on the same dose of lithium.
Approximately 6 months after the sleeve gastrectomy, the patient presented to the emergency department after a fall. She reported feeling lightheaded and nauseous and felt as though her legs gave out. She denied pain or loss of consciousness. Laboratory evaluation in the emergency department showed a urinalysis that was negative for glucose with a specific gravity of 1.010, negative for blood, positive for nitrite and leukocyte esterase, and was positive for bacteria and many squamous epithelial cells. A basic metabolic panel showed sodium of 138 mmol/L, potassium of 3.6 mmol/L, chloride of 102 mmol/L, urea nitrogen of 8 mg/dL, creatinine of 0.92 mg/dL, glucose of 135 mg/dL, and calcium of 10.1 mg/dL. A complete blood count showed a white blood cell count of 6.7 K/µL, hemoglobin of 12.6 g/dL, hematocrit of 38.4%, and platelet count of 299 K/µL. A lithium level was elevated at 1.63 mmol/L. The patient had lost approximately 40 pounds (BMI = 34.9; 14.8% body weight lost) since the time of the sleeve gastrectomy. Differential diagnosis included dehydration and gastrointestinal infection in addition to lithium toxicity.
At this emergency department, the patient was instructed to hold her lithium for 2 days, which the patient did. The patient’s primary physician and a clinical pharmacist reviewed the patient’s record and made the decision to decrease the patient’s lithium dose to 300 mg twice daily.
The patient returned to the emergency department approximately 10 days after the first emergency department visit with difficulty walking and one fall, as well as somnolence. A family friend who brought the patient to the emergency department stated that there was no suspicion of overdose. The emergency department workup revealed a urine toxicology screen that was negative for amphetamine, barbiturate, benzodiazepine, cannabinoid, cocaine, opiates, phencyclidine, and oxycodone. Computed tomography scan of the head showed no acute intracranial abnormalities. Lithium level at that time was 1.10 mmol/L. She was admitted to the hospital for encephalopathy. A differential diagnosis, in addition to lithium toxicity, of dehydration, acute kidney injury, and infection were considered. Psychiatry consultation was obtained, and in collaboration with the primary physician, the decision was made to reduce the patient’s lithium dose to 150 mg bid. The patient has had no further episodes of somnolence, falls, or supratherapeutic lithium level since this dose reduction. A lithium level obtained several months later on the 150 mg bid dose was 1.02 mmol/L. One year following surgery, the patient’s lithium level was 0.53 mmol/L at a weight of 199 pounds (BMI = 30.26; 26.2% body weight lost).
The following table summarizes the patient’s weight loss, creatinine, lithium dosing, and lithium level at baseline, at her presentations to the emergency department, and at follow-up.


A 23-year-old male with a past medical history of Celiac disease, hypothyroidism, homelessness, and prior in-patient treatments for schizophrenia presented to our institution after having amputated his penis, testicles, and most of his scrotum with a serrated knife. This was done in a public area, 20 minutes prior to arrival after taking two ibuprofens. The penile shaft, both testes, and scrotal skin were brought on ice with the patient by the EMT (Figure ).
Upon arrival, the massive bleeding from the penile stump/groin was controlled by applying pressure with epinephrine-socked gauze and the patient was stabilized according to trauma protocol. He denied hallucinations and suicidal ideations. He was withdrawn, had affective flattening, disorganized speech, and difficulties organizing his thoughts. He stated that he was homeless and was undergoing gender affirmation surgery but did not supply details. He did not want his penis to be re-implanted nor his family to be informed. He had dramatically diminished sensation of pain given his injury. His toxicology screen was negative. On examination, his penis was amputated at the base. He was given a third-generation cephalosporin and tetanus-diphtheria-acel pertussis vaccine. The prior psychiatric history of the patient could not be obtained at this time and was later hindered by the family's denial that he suffered from schizophrenia. The ED psychiatrist, the attending trauma surgeon, the reconstructive urologist (DAP), and the administrator-on-call all agreed that the patient was in an acute psychosis and lacked legal capacity to refuse emergent medical treatment. He was taken emergently to the operating room for microsurgical replantation. Aspirin therapy was started, and subcutaneous heparin (SQH) 5000 units TID was initiated. Prior to induction, he again stated that he was a transgender woman, refused reattachment, however, did desire for us to control the bleeding. Surgical details are described in the Appendix .
After the index surgery, the patient was transferred to the surgical intensive care unit for graft perfusion monitoring. To increase penile perfusion, he was started on tadalafil 5 mg daily. Of note, the patient started to experience morning erections of the proximal and partial erections of the distal shaft within 2 weeks from reconstruction.
Postoperative testosterone was 67 ng/dL, and he was started on replacement with testosterone enanthate IM 200 mg every 10 days. Given the contaminated nature of the wound, he was continued on IV antibiotics (switched to oral after 5 days and then continued for 6 days).
Given prior history of hypothyroidism, his thyroid function was evaluated; his thyroid-stimulating hormone (TSH) was >150 uIU/mL and free T4 was 0.3 ng/dL. Levothyroxine was continued and increased to an appropriate dose.
Five days after the index surgery, the patient developed classic venous skin congestion (Figure ) and twice-daily medical leech therapy was initiated (one placed ventrally and once dorsally and allowed to imbibe until they fell off). This initially helped reduced the ecchymosis and hematoma (Figure ); however, it was stopped after 1 week when skin necrosis developed (Figure ). The SPY Fluorescence Imaging System (Stryker) confirmed no perfusion. As the penile skin started to form an eschar, wound care consisted of twice-daily application of MediHoney gel (Derma Sciences) to eschar and Aquacel strips (ConvaTec) to exposed tissues. Daily bedside wound debridement was performed to remove necrotic skin and eschar.
Two weeks after the index surgery, the patient underwent a retrograde urethrogram (RUG) showing no stricture nor extravasation (Figure ); the Foley catheter was removed. He had no postvoid residual and voided without difficulty. Two weeks later, he was transferred to the psychiatric unit with continued local wound care awaiting eventual skin grafting. Prior to transfer to a psychiatric unit, the SQH and aspirin were discontinued and patient was started on pentoxifylline 400 mg TID to improve perfusion to penis.
Approximately 10 weeks after index injury, plastic surgery performed a 0.012”-split-thickness skin graft to the shaft using 110 cm2 from the lateral thigh, sutured in place with 4-0 chromic interrupted sutures. The graft healed without any complications. Once the graft was fully epithelialized, the patient started using a penile pump daily to prevent the loss of penile length.
From arrival in our ED, the psychiatry team was involved. He had been diagnosed with schizophrenia approximately 1 year prior but had not been compliant with his home antipsychotics (risperidone, later switched to paliperidone, and then long-acting injectable paliperidone). He had a family history significant for suicide by multiple paternal relatives and father with bipolar disorder. Before his self-mutilation, he had been a missing person while living undomiciled. He had a history of paranoid and persecutory delusions and suicidal ideations; there was no record of prior gender dysphoria. Daily assessment was performed by psychiatry and urology, and he was told that with improvement, he could be enrolled in a transgender program. After the index surgery, the patient initially stated that he had wanted gender affirmation surgery but had disorganized thoughts and poor understanding of his condition. Initially, he continued to endorse auditory hallucinations commanding him to amputate his genitalia; however, after the psychosis had resolved, he denied gender dysphoria. His antipsychotics were initially risperidone 1 mg twice daily and over the next 2 months up-titrating to 8 mg twice daily and then cross-tapered to clozapine 425 mg daily augmented with lithium 1200 mg daily until therapeutic levels met. He was discharged to a long-term psychiatric facility on lithium 600 mg twice daily, clozapine 150 mg q-AM and 275 mg QHS, and sertraline 150 mg q-AM.
The patient presented for follow-up at 12 months after the index surgery. He was still under psychiatric treatment. He denied being able to ejaculate; however, he was not self-stimulating and denied sexual activity. The patient did endorse tumescence although it was not yet adequate for penetration. Penile sensation was present although reduced. The skin graft was well-healed, the testis was palpable albeit atrophic, and the urethral meatus was patent (Figure ). He was continued on testosterone replacement therapy (testosterone 339.7 ng/dL) and penile vacuum therapy. He was advised to use penile vibratory stimulation to help him reach orgasm if needed. He denied any gender dysphoria nor desire for gender affirmation surgery and identified himself as a heterosexual male.


Our patient is 56-year-old female, a known case of locally advanced breast carcinoma. She was initially managed by chemotherapy followed by modified radical mastectomy then adjuvant radiotherapy and chemotherapy. The patient gave a positive family history of colon cancer; therefore, genetic testing for adenomatous polyposis coli (APC) mutation and surveillance colonoscopy were recommended by genetic counselor. The APC gene mutation came to be positive and as part of the work up for her primary disease, computed tomography (CT) scan of the abdomen performed and showed evidence of 4 cm intraluminal lesion in the ascending colon suggestive of lipoma without bowel obstruction (). As a result of these findings, colonoscopy was performed and it revealed a large smooth intraluminal subepithelial lesion in the ascending colon occupying one third of the lumen circumference (). With the use of biopsy forceps, indentation of the tumor can be seen, which is as known as “pillow sign” and once a biopsy taken, a fatty yellowish material was seen protruding from the biopsy site suggestive of lipoma (). Histopathological examination showed multiple fragment of normal colonic mucosa and fragments of mature adipose tissue consistent with lipoma (, black arrows). Clinically, the patient denied history of abdominal pain, rectal bleeding, changes in her bowel habit and intestinal obstruction symptoms. She denied any personal history of malignancy or lipoma. On physical examination, there was no evidence of any lipomas in particular her scalp, limbs or trunk. Although she has no gastrointestinal symptoms attributed to the lipoma, given that it is large in size and she is in clinical and radiological remission, we referred her for the opinion of colorectal surgery.


A 52-year-old man, with a history of diabetes and hypertension for 9 years, had been diagnosed with ischaemic cardiomyopathy and had undergone coronary artery bypass grafting 1 year earlier. After surgery, there was mild improvement in symptoms but no improvement in LV systolic function, with an LV ejection fraction (LVEF) of 15–20%. Moreover, he continued to have New York Heart Association Class III dyspnoea despite being on adequate medication. An electrocardiogram showed a sinus rhythm heart rate of 78 b.p.m. and left bundle branch block, with a QRS duration (QRSd) of 180 ms.
Cardiac resynchronization therapy was performed under local anaesthesia and intravenous sedation with midazolam and fentanyl. After dissection of the subclavian pocket in the prepectoral fascia, the right ventricular (RV) lead was positioned in the RV apex, followed by a selective CS angiogram with a coronary sheath. A superselective 5 F Headhunter catheter was used to enter the lateral vein. Contrast injection revealed a single small lateral branch with angulation and proximal stenosis (). The branch was wired with a 0.014″ coronary guidewire (RunThrough NS Floppy, Terumo Corporation, Tokyo) and pre-dilated with a 2.5 × 12 mm balloon (Sprinter Legend, Medtronic, MN, USA) at 14 atm for 1 min. An attempt to negotiate an LV lead was not possible due to an unfavourable angle. A venovenous loop was therefore formed to support lead tracking.
The 0.014″ coronary guidewire wire was passed into the distal segment of the lateral vein. The collateral connecting the lateral vein and the middle cardiac vein (MCV) was identified after injection of contrast. The wire was subsequently advanced into the collateral while rotating (clockwise-anticlockwise) to ensure that the tip was freely moving. An adequately sized, less tortuous collateral was identified for wiring. The wire was tracked via the collateral into the MCV and eventually into the CS. It was pushed gently at the wall to form a knuckle and then advanced into the right atrium, and then further into the superior vena cava (). Advancement of the wire did not need any extra-support tools as it was not very tortuous and could be tracked easily. The distal end of this guidewire was caught with an indigenous snare in the high right atrium (). This indigenous snare was prepared using a 6 F Judkins JR3.5 guide, a 0.014″ coronary guidewire (RunThrough NS) and a 2 × 15 mm angioplasty balloon (Sprinter balloon, Medtronic, MN, USA). The second guidewire was passed with the balloon and its distal end was reintroduced into the tip of the guide catheter to form a loop. The balloon was inflated at the tip to entrap and fix the distal end of the coronary guidewire ().
The distal end of the first coronary guidewire was snared, pulled out through the left subclavian vein and externalized through a separate sheath (, Video S1) in the infraclavicular pocket, forming a venovenous loop (). Both ends of the guidewires were placed under the control of the operator. The LV lead could then be advanced easily over the venovenous loop and positioned very stably in the vein ahead of the tortuous segment (). After implantation of the LV lead, the guidewire was carefully pulled out from the leads under fluoroscopic control. Fluoroscopy confirmed good separation of both RV and LV leads (), with the thresholds of the right atrial, RV, and LV leads after implantation being 0.9, 0.7, and 1.5 V, respectively, at pulse widths of 0.4, 0.4, and 1 ms, respectively. The patient was asymptomatic and did not complain of any pain or palpitations during the procedure, nor were any arrhythmias observed. The total fluoroscopy time was 13 minutes and the total volume of contrast agent was 120 mL.
Eleven months later, the patient is being maintained on heart failure medications. He showed significant symptomatic improvement after the procedure. His QRSd had improved to 90 ms with good R wave in V1. Echocardiography showed improved LVEF (25–30%).


A 16-year-old boy suddenly experienced cardiopulmonary arrest while playing soccer, received cardiopulmonary resuscitation and automated external defibrillator (AED) treatment by a bystander and was brought to our hospital after return of spontaneous circulation.
The patient had low exercise tolerance since childhood and was admitted to another hospital at age 12 due to the symptom of chest pain while playing soccer. Transthoracic echocardiographic imaging showed normal left ventricular diffuse wall motion [ejection fraction (EF) = 57%], and formation of a trabeculation with suspected non-compaction at the left ventricular apex. The results of treadmill exercise test were negative and follow-up was performed without treatment. The patient became a high school student and noticed an angina attack twice during exercise, but symptoms were quickly ameliorated by bed rest only. In May 2018, an AED was used by a schoolteacher who suddenly collapsed during soccer. After the third AED operation, return of spontaneous circulation was noted and the patient was brought to our hospital for emergency treatment. On admission, significantly reduced consciousness (Glasgow coma scale; E1V1M2) was noted, and therapeutic hypothermia under ventilator management was promptly introduced for protection of the brain.
The initial workup was 103.2 pg/mL (<18.4) for B-type natriuretic peptide and 0.09 ng/mL (0.00–0.03) for Troponin I. Atrial blood gas analysis were pH 7.233 (7.35–7.45), pCO2 39.0 mmHg (35–48), pO2 101.0 mmHg (83–108), and Lac 93.0 mg/dL (4.5–14.4). Chest X-ray revealed the presence of cardiac enlargement (CTR 58%) and pulmonary oedema. Head computed tomography (CT) indicated unclear medullary boundary on the basilar side and hypoxic encephalopathy was suspected. A 12-lead electrocardiogram indicated sinus rhythm, 2-mm ST depression in the anterolateral leads and borderline QTc (441 ms) (). Transthoracic echocardiographic imaging showed decreased left ventricular diffuse wall motion (EF = 34%), and formation of a trabeculation with suspected non-compaction at the left ventricular apex. As a result of AED analysis, initial waveform indicated ventricular fibrillation ().
When midazolam administration was discontinued on hospital Day 1 for the purpose of assessment of consciousness level, an improvement to GCS E4VtM6 was noted. Therefore, the rewarming period was shortened from 48 to 20 h, the artificial respiration was ceased on hospital Day 2, and the patient was discharged from the intensive care unit on hospital Day 4.
Selective coronary angiography revealed left coronary artery (LCA) defect from the ostium and return of blood flow from the right coronary artery via collateral circulation on hospital Day 7 (). Right coronary artery was well developed, there were no angiectopia or stenosis, and collateral circulation was observed from a posterior descending artery branch to the periphery of the left anterior descending artery (LAD), from a posterolateral branch to the diagonal branch, and from an atrioventricular node branch to left circumflex artery (LCX). Furthermore, a solitary conus branch with a different ostium directly above the right coronary ostium allowed circulation of blood flow to LAD/LCX via Vieussens’ arterial ring. The left main trunk had a blind end at the junction of both (). As a result of collateral circulation, there was no significant stenosis in LAD and LCX revealed by contrast-enhancement.
Coronary angiography CT showed a lack of LCA ostium and no traffic between the left pulmonary artery on hospital Day 9 (). Therefore, LMCAA was diagnosed in the patient. Myocardial perfusion scintigraphy revealed drug-induced blood flow imbalance in the apex and posterior wall and viability of the same site on hospital Day 10. Off-pump coronary artery bypass grafting with two branches (left internal mammary artery–LAD, right internal mammary artery–D1) was performed for the purpose of revascularization on hospital Day 15. Postoperative course was favourable, and coronary angiography CT on postoperative Day 13 revealed favourable bypass graft patency (). Transthoracic echocardiographic imaging on postoperative Day 17 showed dramatically improved left ventricular diffuse wall motion (EF = 59%). After cardiac rehabilitation, the patient was discharged in an ambulatory state on postoperative Day 50. A wearable cardioverter-defibrillator (WCD) was used for approximately 3 months, but no obvious chest symptoms or ventricular arrhythmia were observed, and the patient is currently showing good progress without implantable cardioverter-defibrillator (ICD) implantation.


A 55-year-old male presented with 4 months of left chest wall pain. He noted left nipple discomfort with tenderness to touch. He did not appreciate any enlargement of either breast.
Nine days prior to presentation, he had completed an 8-month course of daily meloxicam 15 mg for knee pain. He was not taking any other prescription, over-the-counter medication, herbal medications or supplements, or tetrahydrocannabinol (THC) containing compounds.
On exam, the patient had exquisite tenderness in the subareolar region of the left breast and palpable breast tissue 4 cm in diameter. The right breast tissue was neither enlarged nor tender.
The differential diagnosis of unilateral gynecomastia and options for further evaluation were discussed with the patient. The patient declined further evaluation but agreed to avoid use of meloxicam and other NSAID medications.
He returned 2 months later and reported that breast tissue tenderness resolved 4 weeks after meloxicam cessation. Clinical examination by the same clinician that had originally seen him revealed no tenderness or palpable breast tissue of either sub-areolar region.


A 59-year-old Caucasian man was admitted at our outpatient Clinic in December 2019 presenting a severe and generalized skin rash characterized by papulo-pustules involving the face, trunk, and limbs. These manifestations were strongly itchy and not responsive to topical steroids and systemic antihistamine drugs, showing a high impact on quality of life. His medical history was positive for hypertension, arthrosis, anxiety, and depression; he also presented a percutaneous nephrostomy for a previous obstructive uropathy (2018) and a percutaneous ileostomy for a previous intestinal obstruction (2017). The patient said that the skin rash began 2 months ago, involving the face and subsequently expanding to the trunk and upper limbs. Dermatologic examination showed follicular papules and pustules, coalescing to form plaques on the face, trunk, and limbs (Fig. ). Routine blood tests were within normal ranges except for peripheral eosinophilia which represented 24.3% of the leukocyte formula, with a 1.91 × 103/μL count of eosinophils (normal value: 0–6%; 0.0–0.45 × 103/μL). An HIV screening was performed with negative results. A 3-mm skin biopsy was sent for histology showing skin with a rich representation of the pilosebaceous appendages, site of severe lympho-granulocytic infiltrate, with an impressive amount of eosinophilic granulocytes, which involved the annexes both superficially and deeply supporting the diagnosis of EPF (Ofuji disease). As shown by the therapeutic algorithm for EPF proposed by Takashi Nomura et al. [], we started oral indomethacin (50 mg). Despite an initial improvement, the patient discontinued the therapy after only 1 week due to gastrointestinal events (diarrhea), manifesting an aggravation of the facial manifestations, with the appearance of new papulo-pustules. We decided to reduce the dosage of oral indomethacin from 50 to 25 mg combining it with topical tacrolimus ointment once a day, observing the spontaneous disappearance of the gastrointestinal symptoms and an excellent response especially on the itch symptom. During the last follow-up (week 8), the patient showed a total absence of itching and an important reduction of facial manifestations (Fig. ).