A 67-year-old male presented to the emergency department with a history of low-grade fever, difficulty swallowing, shortness of breath, and painful swelling of the left lateral neck for 1 day. He had no history of a recent upper respiratory tract infection. His medical history included well-controlled type 2 diabetes mellitus, hypertension, and dyslipidemia. On examination, his temperature was 38.8°C, heart rate was 110 beats/min, and respiratory rate was 24 times per minute. He had mild respiratory distress with inspiratory stridor. There was a tender swollen mass on the left side of the neck, measuring 5 × 6 cm.
The laboratory investigations revealed no leukocytosis (white blood count 9,000 cells/mcL). His thyroid function test showed normal free thyroxine and TSH levels. Computed tomography of the neck with contrast () revealed a large lobulated partially rim-enhancing hypodense lesion size (5.3 × 10 × 15.8 cm) with internal septation involving the retropharyngeal space, orohypopharynx, larynx, and left lobe of the thyroid gland. Findings were compatible with an abscess that caused adjacent airway narrowing and right lateral displacement of the trachea. Lymphadenopathy with cystic necrosis was also detected at bilateral cervical level II and left level III, measuring up to 2.8 cm in the greatest diameter. We cannot demonstrate odontogenic infection and sialadenitis. His provisional diagnosis was DNI, empirically treated with intravenous antibiotics of ceftriaxone and clindamycin. He underwent incision and drainage of the abscess. Only 10 mL of pus was drained from the left paratracheal area because the main component of the mass was the necrotic debris. Necrotic tissue samples were sent for culture and histopathology. Later, culture grew coagulase-negative staphylococci. After a 10-day course of intravenous antibiotics, his clinical signs and symptoms had improved, and he was discharged from the hospital with oral amoxicillin-clavulanic acid. However, PTC was found in the pathology of the necrotic tissue and one left cervical lymph node ().
Extensive preoperative imaging of the thyroidectomy was performed. After incision and drainage, ultrasound showed a 5 cm heterogenic hypoechoic mass with some cystic portion occupying the entire left thyroid lobe (). The color Doppler study showed no hypervascularity. No residual abscess was identified. Multiple enlarged left cervical lymph nodes with cystic necrosis were seen (). Barium swallowing showed a right lateral deviation of the cervical portion of the esophagus due to the compressive effect of a left neck mass. Esophagogram did not show any leak in the cervical esophagus. The fine-needle aspiration biopsy (FNAB) of the right cervical lymph node was performed, and the cytological result was reactive hyperplasia.
He underwent total thyroidectomy with the bilateral central node, left radical, and right selective neck dissection. The pathological result reported a classic PTC with tall cell features (10% of tall cells). The tumor was located on the left lobe, 3.5 × 2.3 × 1.6 cm in size with the presence of extrathyroidal and lymphovascular extension. The resected margin of the tumor was not free. Bilateral cervical lymph nodes showed metastatic PTC, predominantly in the left side. He was subsequently treated with radioactive iodine.


The first patient is a 79 year-old female with a history of 5 prior surgical resections and radiation for a locally recurrent high grade myxofibrosarcoma of the lower extremity. Genomic analysis demonstrated alterations in tumor suppressor genes, including deletions in TP53 (W146fs) and CDKN2A. The tumor was microsatellite stable, with a tumor mutational burden of 3.5 mutations per megabase. She was referred for amputation but opted for an ILI with concurrent pembrolizumab in an attempt to salvage the limb. At presentation she had numerous (at least 12) lesions in the vicinity of her prior operations but no evidence of distant disease (). She received a single dose of pembrolizumab followed by ILI within two weeks and was subsequently maintained on pembrolizumab post-infusion according to the standard dosing schedule. The patient had a partial response of 6 months duration (). All of the lesions that completely regressed continued with a complete response. At 5 months, she developed symptomatic hypothyroidism and pembrolizumab was held. Progression was noted in the lesions that had partially responded before pembrolizumab was resumed.
A biopsy of one of the responding lesions showed a brisk lymphocytic infiltrate (
). The patient went on to other limb salvage treatment when the remaining lesions grew off treatment. She remains alive with disease now 30 months post-ILI.


The second patient is a 77 year-old female who had seven prior surgical resections as well as radiation for multiply recurrent high grade myxofibrosarcoma of the lower extremity. She presented one year after her 7th resection with recurrent disease, a 4.1cm x 2.9cm mass overlying the anterior tibia, a possible adjacent soft-tissue tumor nodule, and metastatic disease in an inguinal lymph node (). Genomic analysis revealed a TP53 (R342*) mutation and ATRX deletion. The tumor was microsatellite stable, with a tumor mutational burden of 0.9 mutations per megabase. She received a single dose of pembrolizumab followed by ILI a week later. She subsequently continued on pembrolizumab for one year. Her response to treatment was rapid with resolution of her palpable inguinal node and softening of her tibial lesion at her 1-month follow up. Both the lesion in the chemotherapy infusion field (tibia) and outside the infusion field (inguinal lymph node) demonstrate ongoing radiologic complete response at two years ().
Compared to the pathologic appearance of the excision specimen a year prior (), a tibial biopsy performed one month after ILI revealed >99% treatment effect (). PD-L1 expression was absent in the post-treatment biopsy, with extensive CD4+ and CD8+ T cell infiltrate, as well as CD163+ macrophage infiltrate ().


Patient presentation
A 64-year-old female presented with a chief complaint of right hip, right anterior groin and right thigh pain. She also reported pain in her left knee and left lower back. Relevant history included a left hip arthrodesis following a vehicle accident approximately 40 years prior. Examination revealed that the patient was able to ambulate very short distances with assistance of a single-point cane and used a scooter for travelling longer distances. When ambulating, the patient did so with an antalgic gait. Conservative pain management for the current presentation had included steroid and local anaesthetic injections in the right hip. Physical examination found that the left hip had no range of motion due to arthrodesis, and no apparent strength of the abductors. While LLD was difficult to accurately assess due to a fixed flexion deformation of the left hip, a valgus deformity in the right knee, severe pelvic obliquity and degenerative scoliosis, the left leg was grossly estimated to be 10-15 mm shorter than the right. No neurologic deficit was present.
Pre-operative radiographs (Figure ) revealed a left-sided hip arthrodesis with the hardware securely in place. Significant pelvic obliquity was also noted on imaging, which further revealed advanced degenerative scoliosis in the lumbar spine and osteoarthritis of the right hip, with a loss of joint space superiorly. While right hip arthroplasty was deemed necessary, it was advised that conversion of the left hip fusion to THA should be completed prior, to optimise the outcome of the right hip THA. The use of computer-assisted navigation was recommended to assist with placement of arthroplasty components.
Surgical procedure
Surgery was performed with the assistance of the Intellijoint HIP® imageless navigation device (Intellijoint Surgical Inc., Kitchener, Ontario, Canada). The patient was placed in the lateral decubitus position, the left hip and lower extremity were prepared and the navigation device was installed as previously described in detail elsewhere []. In brief, the device consists of a camera, a computer laptop workstation and an optical tracker. During use, two surgical screws are inserted into the ipsilateral iliac crest via stab incisions and a pelvic platform is fixed to the screws. The navigation camera sits atop the platform, fixed in place magnetically (Figure , ). The camera captures the position and movement of the optical trackers (Figure ), which can be magnetically attached to a platform fixed to the femur (within the surgical incision) to measure changes in leg length and offset or to surgical instruments (e.g., reamer, cup impactor) to measure reaming angle and acetabular component position. Measurements are displayed on the computer workstation, which sits out of the sterile field but within view of the surgeon, in real time (Figure , ).
Following patient registration and primary incision, the joint was exposed. On inspection, the abductor musculature was atrophied. A trochanteric slide osteotomy was performed via the lateral approach, with previously placed hardware transfixing the joint removed and the femur osteotomized through the fusion mass. The femur was then retracted, and the fused boney mass was reamed to identify the true floor of the acetabulum. Traditionally, we would establish an acetabular target intraoperatively based on identification of the fovea or, in cases where the fovea is not visible, by placing the acetabular component such that its inferior edge is aligned with the base of the ischium. In this case, owing to the altered morphology, these landmarks were not readily visible, necessitating the use of navigation. During acetabular reaming, fixation of the device tracker to the reamer handle allowed for real-time evaluation of reaming angle following each round of reaming. This allowed us to accurately establish a suitable orientation for the new acetabular component consistent with traditional target safe zones. Following preparation of the acetabulum, a 50-mm porous acetabular shell was press fit and stabilised with two screws and a 28-mm constrained liner was then inserted. The orientation of the acetabular component was confirmed with the navigation device as 42° inclination and 10° anteversion by identifying three points on the rim of the shell with a probe/tracker combination, which provided the in situ anteversion and inclination measurements.
Following completion of the acetabular portion of the procedure, attention was turned to the femur, where a modular, non-cemented femoral component was used. Attachment of the navigation tracker to a platform fixed to the proximal femur provided real-time monitoring of leg length and offset during trialing, allowing for accurate measurement of these parameters before final components were implanted. Given the significantly altered morphology of both the femur and acetabulum, and the lack of traditional landmarks used during femoral trialing, the data provided by the navigation system proved invaluable in accurately establishing a biomechanically suitable leg lengthening. Following femoral broaching and trialing of femoral components and confirmation of desired leg length (+10 mm) and offset (17 mm) changes with the navigation system, final femoral components were implanted and the hip was reduced (Figure ).
Total operative time was two hours, with blood loss similar to primary and revision hip arthroplasty (200-300 mL). No transfusion was required.
Follow-up
Six weeks post-surgery, the patient was doing well, with no reported complications. She began to bear weight, as tolerated, at three months. Range of motion was greatly improved, with hip flexion measured at 70°, with 10° of internal rotation and 15° of external rotation. The patient was also able to abduct her left hip passively to 20°. Radiographs at seven months post-procedure showed hardware in stable position (Figure ). The patient subsequently underwent a primary THA on the right side. At 11 months post-fusion takedown, the patient reported ambulating well, using a single cane as an aid. She reported being pain free, had returned to outdoor leisure activities and was, overall, extremely happy with her progress. Final radiographs at 18 months post-fusion takedown showed hardware of the left hip in stable position (Figure ). The patient provided consent for their deidentified information to be used in this case report.


A 57-year-old White female with a history of coronary artery disease (CAD) and disabling angina (CCS III-IV) was diagnosed with COVID-19 in December 2020. Her past medical history included hypertension, dyslipidemia, anxiety, attention-deficit/hyperactivity disorder (ADHD), anemia, neuropathy, bilateral foot drop, osteoarthritis, Ehlers-Danlos syndrome, Budd-Chiari syndrome, and chronic pain. She was hospitalized due to COVID pneumonia and respiratory failure for seven weeks. During her hospital stay, she was intubated and experienced complications including retroperitoneal bleeding and bilateral deep vein thrombosis. She subsequently spent three weeks at a long-term acute care facility, followed by three weeks at a rehabilitation center.
After returning home, she required oxygen (2 L/day) therapy for her chronic respiratory failure with her lung capacity being at 35%-40%. She sustained a chronic cough along with an aching chest, fatigue, “brain fog,” shortness of breath (SOB), and dyspnea upon exertion (DOE). She reported orthostatic symptoms when ambulating and needed to sit down for up to 10 minutes to recover to baseline. The exact POTS diagnosis date is unknown. Five months after her acute phase, the patient was referred for her symptoms to an EECP treatment center for a modified abbreviated regimen of 15, one-hour treatment sessions (three times a week for five weeks) based on previous data []. Her pretreatment blood pressure on day one of EECP treatment was 95/67 mmHg and posttreatment blood pressure upon completion of therapy on day 15 was 105/57 mmHg.
After nine, one-hour treatments of EECP over three weeks, the patient reported an improvement in chest pain, SOB/DOE, and fatigue. She indicated that although she continued to experience symptoms, they were less frequent and less severe and occur for a shorter duration.
Upon completion of 15 sessions, the patient reported having less “brain fog” and feeling “amazing,” with the ability to perform physical and mental tasks she was incapable of prior to EECP. The patient reported that she was using her cane less and was able to walk from one side of the grocery store to the other, which she was unable to do before initiating EECP. This was in line with her six-minute walk test distance improving from 910 ft at baseline to 995 ft post-EECP. Her Patient-Reported Outcome Measurement Information System (PROMIS) Fatigue score decreased from moderate (score = 15) to mild (score = 12) fatigue. Her CCS class decreased by one, and her Duke Activity Status Index (DASI) score improved from 0 to 15.45. Her Seattle Angina Questionnaire and Rose Dyspnea Scale scores remained relatively unchanged.


A 28-year-old male visited our hospital because his mother has been diagnosed with FAP with a pathological variant of the APC gene. The APC gene in his mother was screened using the protein truncation test (PTT), and abnormal bands were found in segments of the APC gene (codons 658–1283).
Total colonoscopy showed that he had more than 100 polyps distributed throughout the colorectum (Fig. ). Further examination revealed that he also possessed the pathological variant of the APC gene (Exon15 c.2751del (p.Asp917Glufs*38)). After he was diagnosed with FAP, he received information that surgery was currently the only way to prevent the development of colorectal cancer. However, he firmly declined to undergo surgical procedures and decided to have strict follow-up with frequent endoscopic polypectomy to prevent the development of colorectal cancer. At the first endoscopic examination after diagnosis, en bloc resection was performed on the maximum diameter of polyps larger than 20 mm, and in total 52 large polyps were resected. In histological examination, all polyps were tubular adenomas (~ high-grade dysplasia) and not adenocarcinomas.
The second endoscopic polypectomy was performed 4 months later. We found a flat elevated type polyp, 20 mm in size, located in the ascending colon (Fig. a). It was on a fold edge and disrupted the vascular pattern of the normal colonic mucosa. Spraying with indigo carmine revealed that it had an irregular cerebriform surface and was paler than the background mucosa with an adherent mucus cap that was not washable (Fig. b). After endoscopic mucosal resection, histological analysis revealed that there were two lesions in the polyp, SSL (Fig. a) and SSL with dysplasia (Fig. b). SSL showed characteristics of saw-tooth protrusions of the epithelium into the glandular lumen comparable with hyperplastic polyps but focally with a more complex architecture, and T-shaped or L-shaped glands and dilated glands at the base of the lesion just above the muscularis mucosae. Goblet cells reached the base of the lesion. Ki67 staining showed abnormal cell growth in the tissue of SSL and SSL with dysplasia (Fig. c and 3d).


A 73-year-old man presented to our facility with complaints of left upper quadrant pain and twenty-pound weight loss. A computed tomographic (CT) scan of the chest, abdomen, and pelvis revealed a huge upper quadrant complex mass (), whereas a biopsy of the mass revealed high-grade malignant neoplasm. He underwent radical nephrectomy, splenectomy, and resection of the diaphragm. Pathology revealed a 23.5 cm mass with histologic grade 4 renal cell carcinoma (RCC) with sarcomatoid (95%) and focal papillary (1%) features that invaded into the perinephric fat, gerota fascia, spleen, and diaphragm, all with negative margins, translating to a pathological stage 4. On immunohistochemistry, the tumor stained positive for CK 7, PAX-8, and CD-10, whereas AE1/AE3 was focally positive (–E). Three months postnephrectomy, he complained of pleuritic chest pain. A CT scan of the chest revealed abnormal tissue in the inferior posterior left pleural space, and left-sided abdominal and peritoneal lymph nodes. Biopsy revealed sarcomatoid RCC. He received radiation therapy to that region and subsequent chemotherapy with sunitinib and gemcitabine. A repeat CT of the chest, abdomen, and pelvis 6 months after therapy initiation revealed no evidence of disease.
Unfortunately, 9 months after chemotherapy initiation, the patient noted a tender subcutaneous nodule in the left chest wall. CT scan of the chest revealed a new irregular area of pleural soft tissue at the posterior medial aspect of the left lower lobe of the lung (). A positron emission tomography (PET) scan revealed multiple suspicious retrocrural and left aortic lymph nodes with intense 18F-fluorodeoxyglucose (FDG) avidity, in addition to several FDG-avid soft tissue lesions in the left chest and abdominal wall. He underwent radiation to the left abdominal wall and due to his high PD-1 and PD-L1 status he was prescribed pembrolizumab, which he received for only 3 months because of grade 4 immune-mediated pneumonitis. Repeat PET scans showed interval resolution of the previous lesions in the peritoneal and retroperitoneal cavities and the posterior left costophrenic angle, indicating a favorable response to therapy. Recent PET scans after more than 4 years of immunotherapy revealed no evidence of recurrent disease.


The patient was a 50-year-old asymptomatic Chinese woman, who had been followed up for a lung nodule in the past 3 years and was not taking any medications. Chest computed tomography (CT) demonstrated a small round tumor with clear margin, which was 7 mm in diameter, in segment 8 of the right lung (Fig. ), and was diagnosed as a benign lesion. The whole body bone scintigraphy did not detect any bone lesion. The levels of serum tumor markers, including alpha fetal protein (AFP), carcino-embryonic antigen (CEA), β-human chorionic gonadotropin (β-hCG), neuron-specific enolase (NSE), carbohydrate antigen199 (CA199), squamous cell carcinoma antigen, cytokeratin-19-fragment CYFRA21-1 (CA211), cancer antigen 242 (CA242), cancer antigen 153 (CA153) and cancer antigen 125 (CA125) were not elevated. A wedge resection was conducted. Neither pleural adhesion, tumor dissemination, nor an accumulation of pleural effusion was noted during the operation. After ten months post surgery, there is no adverse or unanticipated event happened, and the patient is alive without evidence of recurrence or metastasis.
Macroscopically, an unencapsulated, but well demarcated tumor, measuring 7 mm in diameter, was seen adjacent to visceral pleura. On the sectioning, the tumor was white-gray and jelly-like, with a microcystic change. A small amount of mucus-like fluid came out. Histologically, the tumor was consisted of a sheet of large round cells with microcystic appearance, due to the uni-, multi- and less vacuolated cells. The uni-vacuolated cells resembled mature adipose cells and the multi-vacuolated cells resembled physaliphorous cells of chordoma. Most nuclei were small and round, and placed peripherally, but few of them were larger and in an irregular shape. Apparent nucleoli, atypia or mitosis was not encountered. Myxoid substance was observed both in the cytoplasm of vacuolated cells and microcystic space, which was positively staining for alcian-blue/periodic acid-Schiff staining (AB/PAS), and extracellular matrix was not identified. Neither connective tissue containing blood vessels nor inflammatory cell infiltration was detected in the stroma. Cuffs of lymphocytes were observed at the board. Minimal destruction of the alveolar structure was showed at the periphery of the tumor (Fig. ). Immunohistochemically, the tumor was positive for immunohistochemical markers for CK AE1/AE3, Vimentin, S100, Brachyury, epithelial membrane antigen (EMA) and epidermal growth factor receptor (EGFR). The ratio of Ki-67-positive cells within the tumor was extremely low (1%). Thyroid transcription factor-1 (TTF-1), P63, P53 and GATA-3 were negatively staining (Fig. ). Molecularly, amplification and mutation of EGFR gene were not detected by fluorescence in situ hybridization (FISH) analysis (Fig. ) and the amplification refractory mutation system (ARMS-PCR), respectively.


A 38-year-old Chinese male patient presented to the hospital in March 2019 with severe abdominal pain. Computed tomography (CT) imaging indicated that the patient had a ruptured liver tumor in the lower right lobe of the liver and was experiencing bleeding. The patient had a history of chronic viral hepatitis B (HBV) with no previous antiviral therapy. His mother and brother also had a history of chronic HBV. The patient had no family history of cancer. The patient underwent an emergency liver tumor resection on March 2019. A postoperative pathological diagnosis revealed HCC with tumor thrombus in the vessel (). Hepatic arteriography was performed one month after surgery, and no tumor staining was observed in the liver. In addition, Positron Emission Tomography-CT (PET-CT) indicated no systemic metastasis after resection of the tumor in the lower right lobe of the liver. A time course of the entire case is shown in .


On March 13, 2020, a 47-year-old occasional-smoker man with unremarkable past medical history was admitted to our emergency department in the northeast Iran, with myalgia, dry cough, low-grade fever (37.9 °C), shivering and diaphoresis for the past 5 days. His vital signs were stable. He had no complaint of dyspnea and his O2saturation was 97%. There was no history of suspicious contact to a person with COVID-19. His blood sample results showed leukocytosis (19000 cells/μLiter; normal range 4000-10000 cells/μL) with 75 % neutrophil and 20% lymphocyte. C-reactive protein (CRP) was negative but erythrocyte sedimentation rate (ESR) was 25 mm/hour (normal range< 15 mm/hr).
A non-contrast chest CT was done and showed a wedge-shaped consolidation with air bronchogram in the left upper lobe. Another small pleural based consolidation was also seen in the superior segment of the left lower lobe (). According to his good general condition and lack of dyspnea, he was discharged with outpatient orders and treated for COVID-19 infection by azithromycin for 5 days and chloroquine for 10 days. It should be considered that the diagnosis of COVID-19 was only based on the imaging due to the very shortage of PCR kits of COVID-19 at the beginning of the epidemic in Iran.
During this period, he developed weakness and loss of appetite, with no sign of dyspnea or fever. On the 30th of March, 2020 he fainted at home suddenly and experienced severe pain in left hemithorax. He was referred to the emergency department to rule out the cardiac angina. His electrocardiogram (ECG) and troponin level was normal. We performed the second CT at this time to distinguish any complication or progression of coronavirus infection that was encountered with left pneumothorax. Patchy consolidation with air bronchogram was seen in the left upper and lower lobe and right lower lobe. Patchy ground-glass opacity with superimposed septal thickening (crazy paving pattern) was seen in the right upper lobe too (). His O2 saturation was 98% and no sign of respiratory distress was detected and chronic obstructive pulmonary disease (COPD) was ruled out. The only symptom was a pain in the left hemithorax. The patient refused hospitalization; therefore, we sent the patient home notifying him of the red flags and quarantine protocols. Cap amoxicillin/clavulanic acid 625mg was prescribed for one week.
One week later his general condition got better, and only complained from minimal pain in left hemithorax. We performed an x-ray and saw a little pneumothorax. He was in a good shape with normal O2 saturation, so he was sent home with azithromycin for one month. On May 11, 2020, a follow-up CT represented a small (20×11 mm) loculated pneumothorax and pleural thickening in the medial basal part of the left hemithorax. A subpleural band-like opacity was also seen in the anterior portion of left lower lobe and right upper lobe, and patchy ground-glass opacity was more predominant in the left side (). His general condition was good with O2 saturation of 98% and the dry cough has been resolved.


The patient was a 62-year-old man who presented with a nodule in the right upper lobe on X-ray during a medical examination. He had no significant medical history or medication, and blood chemistry findings were normal, including tumor markers and coagulation tests. Chest computed tomography (CT) showed a nodule with a maximum diameter of 20 mm in the right S1 region without enlargement of the lymph nodes (Fig. A). Contrast-enhanced magnetic resonance imaging of the head showed no brain metastasis. TBB was performed, and small-cell carcinoma was diagnosed. A small amount of hemoptysis was observed after TBB, but the symptoms disappeared the next day, and the patient was discharged from the hospital upon unremarkable findings on X-ray. Since the patient had small-cell lung cancer, positron emission tomography (PET)–CT was scheduled to assess for distant metastasis. On the eighth day after TBB, the patient was admitted to the hospital as an emergency due to 200–300 mL of hemoptysis. Contrast-enhanced CT of the chest showed scattered shadows around the right upper lobe nodule suggestive of hemorrhage (Fig. B). Since hemoptysis was reduced after admission, we continued conservative treatment with hemostatic agents and bronchial artery embolization was not performed. On the 12th day after TBB, SpO2 decreased to 92%, and chest CT showed occlusion of the upper lobe bronchus and the right middle bronchial trunk with hematoma, resulting in complete atelectasis of the right middle and lower lobes (Fig. ). Emergency thoracotomy of the right upper lobe lobectomy (RUL) and lymph node dissection 2a-2 (ND2a-2) was performed. The operation was performed with differential lung ventilation through a double-lumen tube. The hematoma was occluding the right main bronchus and more centrally located than on the preoperative CT image. The hematoma in the airway was too viscous to be aspirated with suction pressure from the intubation tube. All lobes of the right lung were inflated, and achieving deflation was complicated by the obstruction of the right main bronchus. The pulmonary artery was identified from the interlobar space, and the ascending pulmonary artery was ligated. The minor fissure was dissected first, followed by the superior pulmonary vein and truncus pulmonary artery in this order. The right upper lobe was removed by dissecting the right upper bronchus with a scalpel, and the hematoma was carefully pulled out from the dissected end of the bronchus with forceps (Fig. ). A suction tube was inserted into the middle and lower lobe bronchus to aspirate the remaining hematoma. The upper lobe bronchus was closed with six stitches using the Sweet method with 4–0 Prolene sutures. The pathological diagnosis was a large-cell neuroendocrine carcinoma with a maximum diameter of 20 mm and pT1bN0M0 Stage I A2. Chest CT on postoperative day 7 showed no atelectasis and good lung expansion. The patient was discharged uneventfully on postoperative day 10.


An eight-year-old male North African child treated for leukemia was referred to our cardiovascular surgery department for management of a right lateral-cervical mass. The patient did not report any history of tonsillectomy or cervical trauma. The child was being followed for acute lymphoblastic leukemia type B for which he was receiving polychemotherapy. He had central venous ports (chemoports) placed via the right internal jugular vein one month prior to admission. The chemoport was complicated by a venous thrombosis with port chamber infection two weeks after its implantation. The chamber and the central venous catheter were removed, and the child received a double intravenous broad-spectrum antibiotic (Amoxicillin-Clavulanic Acid and Sulfamethoxazole) treatment two weeks before admission. The patient subsequently developed a right latero-cervical mass one day before admission.
Upon physical examination, the child was in good general condition, awake and alert. He was feverish (temperature = 38.9 EC); cardiac and pulmonary examinations were normal. Neurological examination was normal, notably without signs of facial weakness. Otorhinolaryngological examination was normal, ears and nose were clean. The pharynx exam was also normal, notably without erythema, exudates, or pharyngeal oedema. The patient had a right latero-cervical tumefaction (
) painful to palpation, pulsatile and expansive (3 cm-long large axis), with inflammatory signs all around.
He also presented a cutaneous ulceration with a diameter of 5 cm, located under the right clavicle (former location of the implantable chamber), with inflammatory signs all around (
). Complete blood count revealed anaemia with a haemoglobin level of 9.6 g/dL (Normal: 11.9-15 g/dl), thrombocytopenia with platelet levels of 126,000 per mcL (Normal: 150000-400000 mcL), and hyperleukocytosis with a leukocyte count of 22,3 × 10
9/L (Normal: 4.5-14.5 × 10
9/L). The C-reactive protein reached 95 mg/dl. (Normal: < 5 mg/dl).
A cervical ultrasound found a right common carotid artery (CCA) false aneurysm measuring 29-28 mm of axis, with inflammation of the right cervical subcutaneous tissue associated with huge cervical lymphadenopathies. A cervical Angio-Computed Tomography (CT) scan (
) showed a 3 cm pseudoaneurysm of the right CCA beginning 2 cm from its origin and at a distance from the carotid bifurcation, with the presence of multiple cervical lymphadenopathies and an unimpaired cerebral circulation through the polygon of Willis.
Haemocultures were carried out but did not find any isolated pathogens.
The patient was operated on the evening of his admission. The procedure was carried out under general anaesthesia. The surgical incision was a lateral right cervicotomy extended to the sternal fork. First, the CCA was dissected and controlled at its origin and terminated just before the carotid bifurcation (
). An arterial clamping was made on both sides of the mycotic aneurysm, and resection of the latter, and all-around infected tissues, was performed (
). Our first strategy was to use the greater saphenous vein for reparation. However, given the extent of the infection with brittle arterial tissue, the existence of cutaneous fistula (
), and the excellent pulsatile arterial reflux from the CCA, the decision was made to make a simple ligation of the artery to avoid complications, especially graft rupture (
). The immediate surgical procedure was simple, with an alarm clock on the operating table and without neurological deficit.
During the postoperative period, the child was hospitalized in the intensive care unit. Excised local lymph nodes and aneurysm tissues were sent for bacteriological and pathologic examination. The culture of bacteriological samples was negative, a predictable result given the probabilistic antibiotic therapy introduced from the first presentation of symptoms. Repeated haematological analysis showed that his leukocyte levels had dropped to normal values.
The patient made an ordinary recovery and was discharged after two weeks. Low-dose aspirin (75 mg per day) with 6 weeks of oral antibiotics (Amoxicillin/Clavulanate 15 mg/kg every 12 hours) were prescribed. Upon one-year follow-up, the neurological examination was normal, and in particular showed no neurological sequelae.


A 28-year-old woman of African origin, gravida 4 para 3+0 at 28 weeks of amenorrhea was referred to Lacor Hospital from a nearby district hospital due to repeated episodes of loss of consciousness, collapse, and tonic jerking of the limbs. These symptoms had occurred over seven weeks and each would last for a few seconds only. She noted that these episodes were common on standing up just before making any steps. Her care-takers also reported associated incontinece of urine occasionally. She did not have any known chronic illness or contact with Tuberculosis. The past three pregnancies were unremarkable as she did not attend antenatal care clinics and delivered at home with the traditional birth attendant (TBA) due to socioeconomic reasons. During this fourth pregnancy, she had booked for ANC at the district hospital with support from a humanitarian organization. The booking blood pressure at 11 weeks was 125/85 mmHg (taken from the upper limbs). She was started on anticonvulsants; phenytoin 200 mg two times a day for 4 weeks then switched to sodium valproate 800 mg per day for 3 weeks but the symptoms remained unchanged.
On arrival at Lacor Hospital, the carotid, brachial, and radial pulses were undetectable and the BP in the upper limbs was unrecordable. The BP from the ankle was 160/100 and pulse rate of 84 beats/minute. Per Abdomen examination revealed a symphysio-fundal height corresponding to 28 weeks with a fetal heart rate of 140 beats/minute that was regular. The other physical examinations were unremarkable. Investigations done include; echocardiogram which revealed normal cardiac chambers and valves. The ejection fraction was 58.57%. A Doppler scan of the aortic arch, descending aorta to iliac artery and renal artery, and electroencephalography findings were normal. There was thickening of the common carotid artery (CCA) and subclavian artery (SCA) with a 50–60% stenosis on Doppler ( and ). Also, a reverse flow pattern was noted within the vertebral artery referred to as subclavian steal syndrome (). The erythrocyte sedimentation rate (ESR) was 40 mm/1st hour. Antinuclear antibody (ANA) and C-reactive protein (CRP) tests were not avalable at our facility. There was no proteinuria on spot urine dipstick. An obstetric ultrasound scan revealed a single live intrauterine fetus with a heart rate of 138/minute, a normal amniotic fluid index (AFI 11 CM), and normal umbilical artery Doppler velocimetry.
After reviewing the findings, we made a diagnosis of Takayasu's Arteritis with convulsive syncope. The patient was started on nifedipine 20 mg twice daily, with prednisolone 30 mg once a day; anticonvulsants were stopped. She was observed for one week on this treatment, the symptoms subsided and the ankle BP came down to 145/90 mmHg. The patient was discharged, and the dose of prednisolone tapered down to 10 mg daily over 4 weeks. She had a normal vaginal delivery at term to a male baby weighing 4.0kg. During the post-natal review, her BP was recorded as 115/75 mmHg and pulse of 80/minute in the upper limbs and she had no symptoms.


An 86-year-old woman was transferred to the emergency and critical care center of Tokyo Metropolitan Bokutoh Hospital from a nearby general hospital with vital signs indicative of shock. She had been diagnosed with urinary tract infection. She had a medical history of chronic heart failure with pulmonary hypertension and was on home oxygen therapy for chronic respiratory failure.
On initial physical examination at arrival to our facility, she was conscious and oriented with a Glasgow Coma Scale score of 15 and a body temperature of 36.0 °C. On receiving a 0.3γ dose of noradrenaline, her blood pressure, pulse rate, and respiratory rate were maintained at 91/63 mmHg, 90 beats/min, and 24 breaths/min, respectively. Her oxygen saturation was 100% while receiving 10 L/min oxygen through a face mask. On physical examination, her left abdominal and left lumbar areas were tender. Her initial arterial blood gas analysis while on 10 L/min oxygen revealed a pH of 7.441, PaCO2 of 44.6 Â mmHg, PaO2 of 275.5 Â mmHg, HCO3− of 29.9 mmol/L, SaO2 of 93.1%, and lactate of 1.3 mmol/L.
The patient’s urine was negative for nitrite, white blood cells, and bacteria. Serum white blood cell count was 11,900/mm3, platelet count was 17.8 mm3, total bilirubin was 0.46 mg/dl, creatinine was 1.94 mg/dl, and C-reactive protein was 6.92 mg/dL. An unenhanced abdominal computed tomography scan revealed left hydronephrosis, adipose tissue opacity around the left kidney, and deviation of the left ureter to the sciatic foramen (Figs. and ). She was diagnosed with obstructive pyelonephritis associated with septic shock due to a ureterosciatic hernia. She was admitted to our intensive care unit for further treatment with resuscitative fluids, vasopressors, and antibiotics. As the patient was hemodynamically unstable, placement of a ureteral stent was attempted for relieving the herniation-associated structural obstruction. On a retrograde ureteral catheter insertion, the ureteral incarceration reduced, and we were able to place a double-J ureteral stent in situ (Figs. and ).
Initially, she required 0.35γ of noradrenaline and resuscitative extracellular fluids to maintain hemodynamic status. However, the day after the stent placement, she gradually recovered from septic shock and was tapered off vasopressors by day 4. Both blood and urine cultures revealed Escherichia coli with good antimicrobial sensitivity.
She continued to receive antibiotic treatment and underwent rehabilitation, with the goal of discharge and continued outpatient care. Unfortunately, she experienced exacerbation of respiratory failure and died on the 32nd day of hospitalization.


Mrs TN is a 61-year-old woman from
Gauteng Province in South Africa (SA).
Informed consent was obtained from the
patient. She is a patient with RA and at the
time of presentation had been managed
with disease-modifying anti-rheumatic
drugs (DMARDs) including methotrexate
and oral corticosteroids. In addition, she
was enrolled in a biological therapeutic trial
using a selective JAK1 inhibitor. She was not
known to be HIV-infected and her known
comorbid conditions included hypertension
and dyslipidaemia.
She was initially admitted to hospital
in May 2019 with a diagnosis of multi-lobar pneumonia, for which she received
empiric antibiotic therapy and was later
discharged. Subsequent follow-up at the
rheumatology clinic revealed worsening lung
infiltrates, despite a lack of symptoms and
clinical complaints. She was referred to the
pulmonology clinic for further assessment,
and her clinical examination was noted to be
remarkably normal with an oxygen saturation
of 93% on room air. Pulmonary function
testing showed a post bronchodilator forced
expiratory volume in 1 second to forced vital
capacity ratio (FEV1
/FVC) of 0.79, total lung
capacity of 4.78 L (109.26% of predicted) and
a diffusion capacity for carbon monoxide per
alveolar volume (DLCO/VA) of 0.34 nmol/
min/kPa/L (21% of predicted). The low
DLCO may be due to a ventilation/perfusion
(V/Q) mismatch secondary to underlying
lung disease.
Her initial chest radiograph from May
2019 showed 2 discrete solid lung nodules in
the left mid to upper lung zones measuring
15 mm × 17 mm and 17 mm × 19 mm,
respectively. In addition, the left lower
zone revealed an ill-defined area of ground
glass opacification . A repeat chest
radiograph in July 2019 revealed that the
lesions had increased in size .
A
computed tomography (CT) scan of the chest
revealed multiple, well defined, peripheral
and patchy air space opacifications with airbronchograms involving the left lower lobe
and abutting the oblique fissure. This was
associated with pulmonary nodules and left
pleural thickening . Initial
blood results revealed a C-reactive protein
(CRP) of 5 mg/L, erythrocyte sedimentation
rate (ESR) of 20 mm/hr and a normal full
blood count. Sputum investigations were
negative for tuberculosis.
Metastases should be bilateral, and the
evolution and doubling time is unlike
malignancy. In addition, infections,
pulmonary infarctions and vasculitic lesions
could be considered, but the patient’s
normal CRP and ESR were not consistent
with an acute inflammatory state. Owing to
the peripheral nature of these lesions, she
underwent a CT-guided transthoracic lung
biopsy, which revealed fibrous connective
tissue with evidence of chronic necrotising
granulomatous inflammation and fungal
organisms with the morphological features
of Cryptococcus
.
Ziehl-Neelsen staining was negative for
acid- and alcohol-fast bacilli. A diagnosis of
pulmonary cryptococcosis was made.
The JAK1 inhibitor was discontinued and
the patient was started on oral fluconazole at
a dose of 400 mg 12-hourly for the duration
of 1 month. Follow-up chest radiography
revealed partial resolution of the lesions
noted previously .
Fluconazole 200
mg twice daily for 3 months and then 200 mg
daily thereafter was continued. Subsequent
repeat CT imaging done 6 months after
initiation of therapy revealed resolution of
lung nodules and masses, with some residual
fibro-cavitary lung changes remaining in
areas previously involved by mass formation
. The patient has fortunately
maintained a stable clinical condition and
remains well, without deterioration of her
lung functions and her RA has remained
stable.


A 46-year-old Iranian man from Kordestan, Iran, was admitted in Tehran Imam Khomeini hospital complex with complaints of weakness, significant weight loss, and dyspnea during recent 4 months.
He had a history of pancytopenia since 8 months ago, and a diagnosis of primary myelofibrosis was established from his bone marrow biopsy revealing fibrosis. His Janus kinase 2 (JAK2) mutation was negative. He was treated with low-dose prednisolone and danazol since 7 months ago.
His main complaints included anorexia, weight loss about 15 kg, fever, night sweats, and progressive nonproductive coughs during recent 4 months.
He was a smoker, about 20 pack-years, and did not have any previous comorbid diseases. His spleen was resected 4 years ago after an abdominal trauma.
On physical examination he was conscious, ill, and cachectic. Oral temperature was 38.4 °C, and respiratory rate was 22 breaths/minute. He had coarse crackle in lungs. No peripheral lymphadenopathy was detected, but the liver was palpable. Laboratory data revealed pancytopenia, elevated alkaline phosphatase (ALP), erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) (Table ). Viral markers for hepatitis and human immunodeficiency virus (HIV) were negative. His purified protein derivative (PPD) skin test was negative. His sputum smear for mycobacterium was also negative.
On chest computed tomography (CT) scan, diffuse ground-glass opacity of both lungs was seen with centriacinar nodules in upper lobes and bilateral pleural effusion (Fig. )
Abdominopelvic CT revealed enlarged liver and accessory spleen, hypodense infiltrative lesions in both kidneys, multiple paraaortic lymph nodes (up to 12 mm), and severe ascites. Lytic destructive lesions in thoracic vertebra 11 (T11) and T12 were also detected (Fig. ).
Bone marrow aspiration and biopsy were performed; the aspiration was dry tap, and biopsy showed multiple focal fibrosis and aggregation of epithelioid histiocytes forming granuloma. Reticulin staining revealed marked fibrosis in marrow spaces particularly around granuloma (Fig. ). Percutaneous liver lesion biopsy was also performed, which indicated necrotizing granulomatous inflammation (Fig. ).
The patient history and findings of CT scans and tissue biopsies were in favor of disseminated tuberculosis. Therefore, anti-mycobacterium therapy was started including isoniazid, rifampin, ethambutol, and pyrazinamide.
Our patient’s dyspnea and appetite improved over the next 10 days. His blood counts increased, and he was discharged after 2 weeks with white blood count (WBC) of 2.9 (103/μL), hemoglobin (Hb) of 10 (gr/dl), and platelet of 140 (103/μL). His final diagnosis was disseminated tuberculosis with secondary myelofibrosis. The anti-TB medications was continued. In follow-up visits after 2 months, his overall clinical status was improved, with 5-kg weight gain and no fever. His blood counts were near normal, including WBC of 3.9 (103/μL), Hb of 12 (gr/dl), and platelet of 177 (103/μL). Anti-TB medications were continued thereafter.


A healthy 20-year-old Caucasian male with no family history presented with a history of stuttering at age 6 years. The patient had attended an annual 1-week intensive residential speech therapy program for eight consecutive years and continued speech therapy but experienced a poor response.
At age 14, patient sought care for his stuttering due to lack of efficacy from his speech therapy. Aripiprazole, 5 mg per day, was initiated and titrated to 30 mg per day based on his tolerability, to which the patient exhibited a positive response (). He subsequently developed a thirty-pound weight gain over the course of 5 years above his natural growth. The patient was then switched to lurasidone 60 mg per day for 1 year and experienced a thirty-pound weight loss but worsening of his speech to pre-treatment level. Patient requested to return to aripiprazole and was restarted on 30 mg per day with a resultant improvement of his speech again. Topiramate was initiated as aripiprazole was started to prevent weight gain and was proven successful. However, after 6 months of receiving aripiprazole, the patient developed minor involuntary movements of his tongue and was diagnosed as having tardive dyskinesia .
Deutetrabenazine was initiated at 6 mg per day, and then increased over time to 12 mg twice a day with resultant improvement of his tardive dyskinesia and further improvement of his stuttering from moderate to essentially mild on the Abnormal Involuntary Movements Scale (AIMS). The patient continues to tolerate both deutetrabenazine and aripiprazole well.


A 30-year-old primigravida was admitted at 38 weeks of gestation with pain in the abdomen for two hours. On admission, she was afebrile, her pulse rate was 92 beats/minute, blood pressure was 112/74 mmHg, respiratory rate was 14 breaths/minute, and oxygen saturation was 99% in room air. As per institutional protocol, a rapid antigen test for COVID-19 was done, which was positive. The patient was shifted to the COVID-19 positive isolation ward. The patient had conceived spontaneously and had regular antenatal visits. She had no history of fever, cough, myalgia, or any contact history with a positive patient. She had no history of any past medical disorders. Her routine blood investigations were sent, which showed moderate thrombocytopenia (Table ).
Her ultrasound showed a live fetus and examination revealed thick meconium-stained liquor. The patient had a cesarean section under spinal anesthesia. The cesarean section was uneventful, but the patient had postpartum hemorrhage due to uterine atony, which was managed with injection oxytocin, injection carboprost, and injection tranexamic acid. A repeat test was done which showed a hemoglobin level of 6.6 gm/dl and platelet count of 28,000. The patient received six units of random donor platelet concentrates and two units of packed red blood cells transfusion after which her hemoglobin level was 9.2 gm/dl and platelet count was 106,000 cells per mm3. The patient recovered well and did not develop any symptoms of COVID-19 disease on further follow-up.


A 28-year-old primigravida was admitted at 38 weeks four days of gestation with complaints of pain in the abdomen and leaking per vagina for four hours. On examination, she was afebrile, her pulse rate was 98 beats/minute, blood pressure was 110/70 mmHg, and oxygen saturation was 98% in room air. She tested COVID-19 positive by reverse transcription-polymerase chain reaction (RTPCR) assay on a nasopharyngeal swab. The patient had regular antenatal visits and had no history of any medical disorder. She had no history of any symptoms suggestive of COVID-19 disease. Her routine blood investigations were sent, which were as follows (Table ).
The patient was monitored for the progress of labor and delivered a live baby vaginally. She was monitored closely during the postpartum period and recovered well. She did not receive platelet transfusion and did not develop symptoms of COVID-19. She was followed up after 14 days and her platelet count had returned to normal range.


A 28-year-old gravida 3 para 2 living 2 at eight weeks of gestation came to the emergency department with complaints of pain in the abdomen and vomiting. She was afebrile on admission, her pulse rate was 110 beats/minute, blood pressure was 100/70 mmHg, and oxygen saturation was 98% in room air. Her rapid antigen test for COVID-19 was positive and the patient was admitted to the COVID-19 positive isolation area. Her ultrasound revealed a hemoperitoneum with ruptured left tubal ectopic pregnancy. The patient was posted for laparotomy and her routine investigations were sent (Table ).
After initial resuscitation, general anesthesia was induced. Laparotomy was done, and clots of approximately 1.5 liters of blood were removed. Left salpingectomy with right tubal ligation was done. The patient received two units of packed red blood cells, four units of random donor platelet concentrates, and six units of fresh frozen plasma intra-operatively and was shifted to the COVID-19 positive intensive care unit. She was extubated on second post-operative day, and her repeat investigation showed a hemoglobin level of 8.4 gm/dl, platelet count of 118,000 cells per mm3. On post-operative day three, the patient complained of breathlessness and her oxygen saturation dropped to 85% in room air. She was started on non-invasive ventilation to maintain optimum oxygen saturation level. Her repeat investigations were sent, which showed an increasing trend of C-reactive protein (CRP) and lactate dehydrogenase (LDH) levels (Table ).
In the background of continuous oxygen requirement and high D-dimer level, the patient was started on injection remdesivir, injection dexamethasone, and injection enoxaparin. She responded well to the treatment and her oxygen requirement started to decrease after three days and she was maintaining saturation of more than 95% in room air after five days. Remdesivir was stopped after five days and the patient was discharged on post-operative day 10 in a stable condition.


A 28-year-old gravida 2 para 1 living 1 at 39 weeks four days of gestation was admitted with chief complaints of pain in the abdomen for two hours. The patient had tested COVID-19 positive two days earlier, was asymptomatic, and was in home isolation. On admission, the patient was afebrile, her pulse rate was 88 beats/minute, blood pressure was 112/78 mmHg, and oxygen saturation was 99% in room air. The patient was a known case of gestational diabetes mellitus and was on tablet metformin 500 mg twice daily. The patient was in early labor and her routine blood investigations were sent (Table ).
Progress of labor was monitored, and the decision of cesarean section was taken in view of repeated late decelerations on electronic fetal monitoring. A repeat platelet count was done, which was 75,000 cells per mm3. In view of the rapidly declining platelet count, four units of random donor platelet concentrate transfusion were done. The patient had a cesarean section under spinal anesthesia. Intraoperative and postoperative period was uneventful. The patient was discharged after three days and the platelet count before discharge was 130,000 cells per mm3.


Mr H, a 76-year-old retired school teacher, was referred by his general practitioner to the gastroenterology service in mid-2016 with iron deficiency anaemia and weight loss. His significant past medical history included urinary bladder cancer, which was definitively treated by radical cystoprostatectomy and ileal conduit surgery in mid-2015. He was referred to the cardiology services at the end of 2015 due to shortness of breath and had normal results on chest x-ray, electrocardiography and echocardiography and no explanation was found to account for his “breathlessness”. At the presentation to the gastroenterology clinic, he described feeling nauseous, lack of appetite, and a loss of more than 10% of his body weight over the preceding 6 months. He reported feeling dyspneic and lethargic, and passing unformed stools since his operation, but denied any bleeding. Blood tests showed iron deficiency anaemia, low folate level with macrocytosis, low phosphate and borderline low calcium levels and raised parathyroid hormone level. Testing for coeliac disease using anti-tissue transglutaminase IgA antibodies was negative with a normal IgA level. A review of historical test results showed peri-operative anaemia, partly corrected after iron supplementation, and a negative Bowel Cancer Screening Programme stool test result from 2 years ago. Stool cultures, tests for ova, parasites and cysts, and Clostridium difficile toxin were all negative. CT-colonography showed a normal appearance except for some diverticulosis involving the sigmoid colon. Apart from the expected post-operative changes and an incisional hernia affecting the anterior abdominal wall, there was no suggestion of an extracolonic mass lesion. Upper gastrointestinal endoscopy was visibly normal but biopsies were taken from the proximal duodenum.
While awaiting the reporting of the duodenal biopsy and outpatient clinic follow-up, Mr H progressively became cachectic with increasing nausea and anorexia and started showing some cognitive changes. He was admitted to the hospital, as he could hardly tolerate any oral intake. There was tachypnoea, disorientation and apathy, and he was struggling to make any coherent conversation. On admission, he was found to be in profound metabolic acidosis with hyper-chloremia, and significantly reduced renal function. The additional blood results are shown in Table .
Ultrasound scan of the abdomen showed both kidneys to be of normal appearance, with no hydronephrosis or hydroureter, and a CT scan of the head revealed no overt abnormalities. A repeat CT scan of the chest, abdomen and pelvis, with contrast, showed normal lung fields, no filling defects in the pulmonary arteries, normal configuration of the ileal conduit with the parastomal hernia and a midline incisional hernia containing small and large bowel loops but no features of incarceration or strangulation. No enhancing mass lesion was noted.
The patient was started on oral sodium bicarbonate replacement 500mg three times a day, to correct his metabolic acidosis. Within a few days the clinical picture rapidly changed; cognitive changes and anorexia improved, tachypnoea settled, the patient started tolerating oral intake and began to mobilise independently. Correction of electrolytes like phosphate, magnesium and potassium was done as needed with regular monitoring for re-feeding changes. The hyperchloremic metabolic acidosis was corrected with the continued use of sodium bicarbonate tablets. He was discharged, having significantly improved, after a hospital stay of three weeks.
While he was an inpatient, the duodenal biopsy was reported as showing villous atrophy with moderate blunting of the villi. The lamina propria showed a moderately dense, mixed acute and chronic inflammatory cell infiltrate with some crypt hyperplasia. The overlying surface epithelium also showed infiltration by mixed inflammatory cells including scattered lymphocytes and neutrophil polymorphs. No dysplasia or malignancy was found. However, given the details of the case further serological and clinical correlation was advised. Repeat tests for anti-tissue transglutaminase IgA antibodies and anti-endomysial IgA antibodies were negative. HLA- DQ2 and DR8 genotypes were negative, thus coeliac disease was conclusively ruled out. Given the cognitive changes noted at admission, the possibility of Whipple’s disease was considered and Periodic acid-Schiff and Periodic acid-Schiff-diastase stains were carried out. However, no convincing Periodic acid-Schiff (PAS)-positive macrophages were identified and Tropheryma whipplei DNA PCR was negative on both blood and biopsy samples. As the cognitive changes resolved rapidly after correction of the metabolic acidosis, the cerebrospinal fluid study was not pursued. HIV testing was negative. The serum immunoglobulin electrophoresis showed normal levels of immunoglobulin IgG, IgM and IgA. A review of his medication history revealed none of the drugs associated with drug-induced villous atrophy, were involved at any point. While he had had previous small bowel surgery, there were no real blind loops and his serum folate was low so SIBO was not considered as the cause here. Whilst the antinuclear antibody test was positive, extractable nuclear antigen (ENA) antibodies and anti-double-stranded DNA antibodies were both negative. Anti-enterocyte IgG and anti-goblet-cell IgG antibodies were also negative.
Following discharge from hospital and with continued clinical improvement from correction of his acidosis with sodium bicarbonate tablets, his nausea and anorexia resolved, his oral intake increased and he started to gain weight. At follow-up clinic review, given that we did not feel we had a full explanation of the villous atrophy, we arranged for repeat duodenal biopsies. The histology showed an improvement in the villous blunting, with some crypt elongation remaining but no increase in intraepithelial lymphocytes.
When the patient was seen in the clinic later that year, he reported feeling the best he had felt since his original cystoprostatectomy, and the anaemia and weight loss had fully corrected. A further upper gastrointestinal endoscopy was arranged, two years since the first one, and duodenal biopsies showed nearly normal duodenal mucosa, with villous architecture within normal limits and certainly improved in comparison with his previous biopsies.
The basic therapeutic intervention that had been instituted in this patient was the sodium bicarbonate tablets: by correcting the metabolic acidosis, his clinical condition dramatically improved and his appetite increased. We postulate that this patient’s villous atrophy was driven by a significant reduction in oral food intake and by reversing the starvation, the villous atrophy reverted as shown on longitudinal follow-up with serial duodenal biopsies. While this phenomenon has been demonstrated in animal experiments and in malnutrition in paediatric populations, we did not come across reported cases in adult populations where the recovery of the villous atrophy has been demonstrated with longitudinal follow-up.


A 30-year-old, 75 kg, ASA 3, male patient presented for repair of paraesophageal hernia. His medical history was significant for Leigh syndrome due to MRSP34 deficiency which had resulted in progressive neurological deterioration. The neurological symptoms were marked by cognitive dysfunction, tremors, peripheral neuropathy, and balance problems for which he was taking biotin, riboflavin, ubiquinone, vitamin E, and lipoic acid. His baseline venous lactic acid was 2-3 mmol/L. Preoperative arterial blood gas (ABG) showed pH 7.34, partial pressure of carbon dioxide (PaCO2) 39.8 mmHg, partial pressure of oxygen (PaO2) 97 mmHg, bicarbonate (HCO3) 21.9 mmol/L, and base excess −3. Preoperative transthoracic echocardiography (TTE) revealed a left ventricular ejection fraction (LVEF) of 65% and no valvular dysfunction. He underwent a robotic transabdominal paraesophageal hernia repair under general anesthesia, requiring modest doses of norepinephrine and vasopressin. Intraoperatively, he received 3 L of plasmalyte, 750 mL of 5% albumin, and 100 meq sodium bicarbonate. Estimated blood loss (EBL) was <50 mL. Postoperatively, patient was hemodynamically stable, extubated, and was recovering in the intensive care unit (ICU) for close monitoring given the known history of mitochondrial disease.
On postoperative day (POD) 1, a chest X-ray demonstrated a new pneumomediastinum, and he was taken back to the operating room for repair of a suspected esophageal perforation. During the repair of the esophageal perforation, the first intraoperative arterial blood gas (ABG) revealed a pH of 7.17, PaCO2 22.8 mmHg, HCO3 8.3 mmol/L, base deficit 18, and lactate 9.7 mmol/L. Throughout the case, it became increasingly difficult to maintain a mean arterial pressure (MAP) > 65 mmHg despite fluid resuscitation and escalating infusions of norepinephrine and vasopressin. The acidosis also continued to worsen with a pH 7.07. At this point, tromethamine (THAM) infusion was initiated, and 10% dextrose was substituted for plasmalyte as maintenance intravenous fluid. This resulted in an improvement in ABG (pH of 7.2, base deficit of 13 mmol/L, and HCO3 of 13 mmol/L), but the lactate level remained the same, and he continued to require significant vasopressor support. He received 5 L of plasmalyte and 750 ml of 5% albumin during the procedure. The EBL was 50 ml with 2.5 L urine output.
The patient was transported intubated and sedated to the surgical intensive care unit where high-volume continuous venovenous hemodiafiltration (CVVHDF) was promptly initiated (effluent dose 35 mL/kg/hr) for acute acidosis despite adequate urine output. He remained hypotensive with MAPs of 45–60 mmHg despite norepinephrine 0.32 mcg/kg/min and vasopressin 0.04 U/min. His acidosis continued to worsen with arterial pH 7.05 and lactate 16 mmol/L. A TTE was done which showed a LVEF of 35%, with diffuse hypokinesis. The right ventricle was mildly dilated, but with normal systolic function. There was moderate to severe tricuspid regurgitation (calculated right ventricular systolic pressure (RSVP) 33.5 mmHg) and mild to moderate mitral regurgitation. An epinephrine infusion was added to improve inotropy.
Unfortunately, at this time, the patient also developed severe hypoxic respiratory failure, with chest X-ray showing bilateral pulmonary infiltrates despite mechanical ventilatory support with synchronized intermittent mandatory ventilation with pressure support with tidal volume 480 ml, positive end expiratory pressure 10, pressure support of 10 cm H2O, and fractional inspired concentration of oxygen at 60%. ABG on these settings revealed pH 7.04, PaCO2 28 mmHg, PaO2 67 mmHg, HCO3 7.6 mmol/L, and base deficit of 21.7. Due to persistent hypoxemia, progressive acidosis, and hypotension, the ECMO team was consulted, and they decided to proceed with VA-ECMO. The patient was cannulated with 23 Fr venous and 17 Fr arterial cannulae (Abbott Cardiovascular, Plymouth, Minnesota, USA) in the right femoral vessels and with a 9Fr reperfusion cannula to the superficial femoral artery (Maquet Cardiopulmonary AG, Hirrlingen, Germany). Dextrose infusion was increased to 6 mg/kg/min, and total parenteral nutrition was started. Insulin infusion was used to maintain the blood glucose level in the range of 100–120 mg/dL.
Within two hours of VA-ECMO initiation, the patient's lactic acidosis improved (). By POD 5, his lactate was 1.5 mmol/L, and by POD 6, he no longer required vasopressors, inotropes, or CVVHDF (). VA-ECMO was discontinued without complications. The patient required tracheostomy due to slowly resolving hypoxemic respiratory failure. TTE on POD 15 revealed normal size and function of all cardiac chambers. LVEF was 70%, with no significant valvular abnormality. He was successfully discharged.


We report a case of a 13-year-old Yemeni female who was referred to the rheumatology clinic with a history of joint pains, bone pains, and bone deformity for 7 years. She was born via spontaneous vaginal delivery without complication to consanguineous parents of Yemeni origin. She is the third born in her family. There is no history of joint disorders in the family. Joint pains and bone pains started at 6 years of age. This was followed by easy fatigability and swelling of the proximal (PIP) and distal interphalangeal joints (DIP), followed by the elbow joints. This was accompanied by pain in the left hip, causing her to limp, and finally the inability to walk.
Initially, at the age of 9 years, she presented to the general practitioner with joint and bone pains. She was diagnosed with juvenile idiopathic arthritis and treated with non steroidal anti-inflammaroty drugs (NSAIDS). However, her condition kept deteriorating despite the NSAIDS for 4 years, and she was referred to a pediatric rheumatologist for further management. Her weight and height on presentation were below the third percentile, and upper-to-lower body segment ratio was 1. On examination, the patient was in generally fair condition. She had marked prominence (swelling) of proximal (PIP) and distal interphalangeal joints (DIP) bilaterally, which were non-erythematous and non-tender (Figs. ,). However, there was limited mobility of the joints. Swelling of the knee and elbow joints bilaterally was also noted. She also had scoliosis on presentation.
She was prepubertal at 13 years of age. Neurological examination did not reveal any abnormal findings. Radiographs of the hands and spine were requested.


A 64-year-old female was admitted to our hospital due to dysphagia and chest pain persisting for 2 months. The medical history of the patient and her family was not significant. She presented herself at our thoracic department. Results of physical examination were normal. Gastroscopy revealed an esophageal submucosal protrusion with a smooth surface. This mass was about 25 cm from the superior incisor (). We didn't perform endoscopic biopsy because of its benign features on endoscopic imaging.
The chest computed tomography showed a round high-density shadow on the right edge of the esophagus at the level of the azygos vein (, ).
After preoperative preparation, the complete thoracoscopic enucleation was performed under double-lumen endotracheal intubation of anesthesia. The patient was placed in left lateral decubitus position about 450 frontal incline (semi prone position). The screen was in front of the patient. The surgeon and the assistant surgeon were at the back of the patient. Three trocarts were used. The first trocart of 1 cm in diameter was in the sixth intercostal space at the anterior axillary line for the camera. The second trocart of 1 cm in diameter was in the seventh intercostal space at the posterior axillary line for instruments and devices. The third trocart of 0.5 cm in diameter was in the fourth intercostal space at the middle axillary line for instruments ().
We didn't use artificial pneumothorax by CO2 insufflation. The thoracoscopic visualisation demonstrated an esophageal intramural tumor at the level of the Azygos vein ().
The mediastinal pleura was incised longitudinally above the tumor. The esophageal segment containing the tumor was exposed. The muscle of the esophagus was cut to expose the tumor that has a white, grey color and smooth surface. The tumor was sutured with a vicryl 2.0 to facilitate pulling up the tumor. The tumor was isolated and enucleated completely out of the mucosa. We performed the enucleation of the tumor without cutting the Azygos vein (, ).
Then we injected 50 ml of air into the esophageal lumen but no bubble of air at the bed of the tumor was recognized. The muscular layer of the esophagus was closed by interrupted sutures of vicryl 3.0. The mediastinal pleura was also sutured (). The tumor was placed in an endo-bag and removed out of the thoracic cavity through the incision at the eighth intercostal space at the posterior axillary line. We washed the thoracic cavity by crystal solution. After checking the bleeding at the sites of trocars, a chest tube 28F in size was placed into the pleural space through the incision for the camera. All incisions were sutured by silk. The procedure lasted for 2 h. There were no perioperative complications. The blood loss was minimal.
After the operation, the patient was transferred to the surgery intensive care unit. 6 h later, she was transferred to our thoracic surgery department. On the first postoperative day, the patient swallowed 10 ml of methylene blue solution. There wasn't any leakage of the methylene blue in the chest drainage. On the same day, the chest x-ray was normal. Therefore, the chest tube was removed. The patient ate soft food on the third postoperative day and normal diet from the fourth postoperative day. She discharged from the hospital on the fifth postoperative day. The pathological result is leiomyoma (). The immune-histochemical staining was positive for desmin and smooth muscle actin. Furthermore, it was negative for CD 34 and CD 117.
During the follow up three months later, the patient didn't have the dysphagia and the vague chest pain. The barium swallowing x-ray was normal ().The patient was satisfied after operation. She stated that she would recommend other patients for the complete thoracoscopic enucleation of the esophageal leiomyoma.
This case was operated by Dr. Trong Vu Than who is the chief of the thoracic surgery department of Da Nang hospital in Vietnam. He is a senior thoracic surgeon who has a vast amount of experience in video assisted thoracic surgery.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images.


A 71-year-old male patient presented to the emergency department (ED) with asthenia, loss of appetite, and nonquantified weight loss evolving, at least, over the last six months. Associated with these constitutional symptoms, the patient also mentioned dyspnea for progressively smaller efforts, which impacted the patient’s autonomy and quality of life. At the observation, he was only able to walk from the bed to a chair, with grade III functional capacity according to the New York Heart Association (NYHA). More recently, he presented episodes of paroxysmal nocturnal dyspnea associated with orthopnea that appeared for the first time three months prior to observation, with complaints of occasional dysphagia for solid and liquid food. No other neurological symptoms were present at this time. At this point, the patient consulted a cardiologist, and a transthoracic echocardiogram revealed a left ventricular dysfunction with a left ventricular ejection fraction (LVEF) of 36%. Coronary angiography was also performed, which showed no significant coronary artery lesions. One week prior to observation in ED, the patient started complaining of dyspnea at rest with a dry cough. A diagnosis of respiratory infection was assumed, and antibiotic treatment with levofloxacin was started, with no clinically relevant improvements, which lead the patient to observation in our ED. During disease progression, there was no history of fever, syncope, chest pain, or palpitations.
Regarding past medical history, he was a previous smoker, had permanent atrial fibrillation, was hypocoagulated with apixaban, and had stage IIIA chronic kidney disease according to the "Kidney Disease: Improving Global Outcomes" (KDIGO) classification.
On physical examination, the patient had a peripheral oxygen saturation of 92%, with normal values for blood pressure and heart rate. Jugular venous pressure was present at 45º. Cardiac auscultation was arrhythmic, and pulmonary auscultation revealed inspiratory crackles in the lower half of both lungs. There was no peripheral edema.
A 12-lead electrocardiogram (EKG) was obtained, which showed atrial fibrillation, with a heart rate of 49 bpm, and a complete left bundle branch block (Figure ).
An arterial blood gas test revealed a hypoxemic respiratory failure, with a partial oxygen pressure of 55 mmHg. The remaining blood tests showed a hypernatremia of 149 mmol/L and an elevated B-type natriuretic peptide (BNP) of 754.9 pg/mL. The remaining results of the blood tests can be seen in Table .
A thoracic computerized tomography (CT) scan was performed and showed a significant cardiomegaly, right pleural effusion, and ground-glass opacities in both lungs (Figure ). A cranioencephalic CT scan was also performed, revealing a cerebral atrophy with a subcortical pattern (Figure ).
The patient was admitted for study and symptomatic treatment, assuming the diagnosis of acute heart failure with reduced ejection fraction (HFrEF) of unknown etiology, associated with constitutional symptoms and occasional dysphagia.
We initiated treatment with intravenous furosemide and started prognostic modifying drugs for HFrEF, with progressive improvement of the congestive symptoms. However, he developed a hypercapnic respiratory insufficiency. Respiratory function tests were made, revealing a restrictive ventilatory pattern. A cervical CT scan was also made, revealing no significant alterations. At this point, the suspicion of a neuromuscular disease began to rise, and an electromyography test was performed, showing signs of active denervation and reinnervation in three body segments (cranial, cervical, and lumbar). With a more thorough clinical interview, a family history of DM1 was identified (Figure ), and genetic testing for this disease revealed a pathogenic expansion of the DMPK gene, with a trinucleotide cytosine-thymine-guanine (CTG) repeat size of 312. A diagnostic of DM1 was made in this patient. After stabilization, the patient was discharged to the outpatient setting for follow-up. In the outpatient setting, a cardiac magnetic resonance was made, which identified a dilated cardiomyopathy and alterations suggestive of previous ischemic events in the medial third of the inferior wall of the left ventricle and in the septal wall. Cardiac catheterization was repeated, maintaining no significant coronary lesions. Regarding the findings in the complementary examinations, a diagnostic of a dilated cardiomyopathy in the context of DM1 was made.


The patient was a woman in her fifties who sustained a head injury as a result of DV by her husband. She was transported by ambulance to the emergency department of our hospital. Her medical history included panic disorder and sleep disorder. Computed tomography (CT) revealed a right orbital floor fracture and sinusitis extending into the inferior of the orbit and into the maxillary and ethmoidal sinuses (Fig. A). The orbital floor fracture was judged by a radiologist to be an old fracture. An emergency physician explained to the husband the need for her to get an eye checkup for ocular symptoms because of the old blowout fracture.
Forty-three days after the first visit to our hospital, she was hit again by the husband but continued to endure eye pain at home by using over-the-counter drugs, specifically analgesics, eye drops, and eye ointments. The sharp right eye pain and periorbital swelling gradually deteriorated, and pus began to appear from the right eye. Twenty-eight days following second violence, she accepted to undergo ophthalmological examination at our hospital. On presentation to us, the ocular surface of the right eye was covered with pus. Swelling of the upper and lower eyelids made eyelid closure impossible, and exophthalmos, severe corneal ulcer, panophthalmitis, and no light perception were observed (Fig. B). Although antianxiety and antipsychotic drugs were used under the direction of a psychiatrist as she started having a panic attack in the consultation room, ultrasonography and electroretinography could not be carried out. There was no obvious bruising around the eye and no tear on the ocular surface at that time. Head CT showed the old blowout fracture and chronic sinusitis as observed previously, but also orbital cellulitis and maxilla fracture that had not been previously detected. Orbital CT also showed that the shape of eyeball was maintained and that there was neither foreign body nor abscess in the orbit. It was noteworthy that there was no new blowout fracture. These findings raised concerns that orbital cellulitis could progress to intracranial infection unless intensive treatment was started. These potential risks were explained to her and the husband, but consent to immediate admission could not be obtained. She also strongly insisted on returning to the marital home and did so. Gram stain of pus sample obtained from the ocular surface showed gram-positive cocci, gram-positive bacilli, and gram-negative bacilli. In addition, Mitis. streptococcus and P. micra were isolated on anaerobic culture, with gamma-streptococci on aerobic culture.
The patient returned to our hospital at thirty-five days following second violence due to intolerable pain. As it seemed the topical and oral levofloxacin hydrate (Cravit®) had been ineffective, we recommended enucleation for the purposes of reducing the pain as quickly as possible and preventing orbital cellulitis from progressing to encephalitis. After obtaining her consent, surgical drainage and irrigation of the sinuses and enucleation were performed on the same day. Examination of the enucleated eyeball confirmed large abscess in the anterior chamber and infiltration of neutrophils into the interstitium of the cornea and conjunctiva (Fig. C). From this pathological result, we considered that orbital cellulitis had progressed to panophthalmitis. Gram stain of a pus sample from the maxillary paranasal sinus at surgical drainage yielded gram-positive cocci, gram-positive bacilli, gram-negative bacilli, and leucocytes. Again, M. streptococcus and P. micra were isolated on anaerobic culture.
Postoperatively, following intervention by the police and medical social workers, the psychiatrist proposed that the patient be transferred to the psychiatric hospital secretly and be kept under isolation from the husband. Nevertheless, she showed strong intention to return home with the husband and did so. After the pain resolved, she was referred to the department of plastic surgery at another university hospital to receive a prosthetic eye.


A female infant was born at 27 weeks of gestation to a 33-year-old, Gravida 8, Para 5 mother. The pregnancy was complicated by subchorionic hemorrhage at 11 weeks, rupture of amniotic membranes at 24 weeks gestation, and intrauterine growth restriction with absent end-diastolic flow of the umbilical artery. Doppler scans and a poor biophysical profile (2/8) at 27 weeks highlighted the need for an urgent cesarean section.
The patient received a complete two-dose course of betamethasone at 24 weeks and a single rescue dose before the C-section at 27 weeks. The birth weight was 680 g (13th percentile), and the head circumference was 22 cm (6th percentile). The infant received delayed cord clamping for 1 min, and her 5-min Apgar score was 8. Noninvasive intermittent positive pressure (NIPPV) was initiated in the delivery room, which was continued on admission to the NICU.
A chest X-ray showed radiographic features of RDS (Fig. ). There was an increase in the oxygen and ventilatory requirements. The infant met the institutional criteria for surfactant delivery via LISA. In our institute, the surfactant is administered via the “Hobart method”, which utilizes a semirigid 16-G catheter that is placed into the trachea without the use of Magill forceps. Bovine lipid extract surfactant (BLES®, 5 ml/kg) was successfully instilled.
The catheter was prepared by removing the introducer needle and marking the desired estimated depth. The catheter tip was slightly curved to mimic the natural airway anatomy to facilitate the insertion past the vocal cords. The infant was premedicated with atropine and fentanyl.
The procedure was performed by a neonatal physician trainee who was certified to perform LISA. The vocal cords could easily be visualized using a laryngoscope. Despite easy visualization, an attempt to insert the catheter between the vocal cords to the desired depth failed due to unexpected resistance. Given that the angiocath tip was passed beyond the vocal cords, a decision was made to start the instillation of surfactant. However, resistance was encountered during instillation, which prevented the administration of surfactant. At this point, a second visualization with the laryngoscope was conducted that confirmed that the catheter was through the vocal cords. The angiocath was then withdrawn by 0.5 cm.
A second attempt to deliver surfactant was performed, and this time, it was successfully instilled without any resistance. Subtle subcutaneous neck swelling was noted after the procedure, vital signs were normal, and no bleeding or clinical deterioration was observed. The orogastric tube was aspirated, and no surfactant was retrieved, thereby confirming that there was no inadvertent intragastric delivery of surfactant. The oxygen could be weaned down from 30 to 21% by 45 min after the procedure, and the ventilatory parameters were weaned.
Three hours after the procedure, the infant became hypotensive. A pneumothorax was suspected, so chest X-ray was ordered and revealed a pneumomediastinum (Fig. ). The air leak was localized to the anterior mediastinum, with a linear radiolucency showing a tract to the anterior aspect of the trachea close to the level of the vocal cords, suggesting a high tracheal perforation. Air tracking up to the soft tissue in the neck region was noted, suggesting subcutaneous emphysema.
The infant required prompt management of hypotension, which was addressed with a fluid bolus of normal saline, and needed initiation of dopamine that was ultimately weaned off 8 h later. The pneumomediastinum was conservatively managed with close monitoring of the clinical condition and switching from NIPPV to nasal continuous positive airway pressure (nCPAP) and decreasing positive end-expiratory pressure (PEEP).
The infant remained stable from a respiratory perspective on a low level of noninvasive support. Follow-up chest X-ray revealed a gradual resolution of the pneumomediastinum, and a complete resolution of the radiographic findings was noted by day 10 of life. There was no need for a further dose of surfactant or intubation for mechanical ventilation.
The newborn was transitioned from nCPAP to a high-flow nasal cannula at 32 weeks. Cranial ultrasound performed on day 3 showed a grade 2 germinal matrix hemorrhage on the right side and a periventricular hemorrhage on the left side.
Subsequent cranial ultrasound examinations demonstrated the evolution of periventricular hemorrhage into cystic lesions. The infant had a brain MRI at 32 weeks that showed similar findings. The newborn also had an episode of late-onset sepsis at 20 days of life that was successfully treated with antibiotics. Enteral nutrition was gradually established. The infant was completely off respiratory support by day 50 of life at the corrected gestational age of 34 plus 1 day and therefore did not develop bronchopulmonary dysplasia. The infant was discharged home at 40 weeks’ gestation.


A 65-year-old female presented to the surgery clinic with a six-week history of a pruritic, bleeding scalp mass in the left parietal-temporal region. The lesion had been asymptomatic since childhood but had grown over this six-week period. Examination of the scalp revealed a raised, fungated lesion measuring 4.7 x 4.0 cm with overlying alopecia secondary to the lesion (Figure ). The irregular mass was erythematous with telangiectasias and several areas of punctate bleeding and erosion. The mass was subsequently resected via wide en bloc excision with split-thickness skin graft closing utilizing tissue from the patient's left medial thigh (Figure ). Biopsy revealed a large basal cell carcinoma with predominantly nodular and focal infiltrating features occurring concurrently with an adjacent sebaceous neoplasm (Figures and 4). The latter was comprised of well-circumscribed nodules of basaloid cells with mild to focal moderate cytologic atypia and frequent mitoses with an absence of peripheral palisading and retraction artifact favoring a sebaceoma.


A 34-year-old Chinese male visited his local tertiary hospital due to progressively worsening occipital headache for the past 4 days, without showing any other symptoms of fever, projectile vomiting and so on. His medical history showed that he had had cryptococcal meningitis and HIV infection for about 1 year, and he had been receiving (with good compliance) fluconazole and tenofovir-lamivudine-dolutegravir based antiretroviral therapy (ART) during the same time period. The levels of the inflammatory markers were normal (Leukocytes 5.61*109 cells/mL, C-reactive protein 7 mg/dL). His neurological signs showed no abnormal indications. Head Computed Tomography (CT) scan revealed no abnormalities. The lumber puncture was performed and the test result of CSF is shown in . It shows rising levels of OP, lymphocytes and protein in CSF. The cryptococcus capsular antigen test result of CSF was positive but the results for the gram staining and culture study were negative. The patient was diagnosed with cryptococcal meningitis and was given fluconazole (400 mg, QD) continuously and TMP-SMX (0.48, QD) for 1 week by the doctor at the local hospital. However, his headache problem was not resolved. Then he came to our institution, the Infectious Disease Department of the First Affiliated Hospital, School of Medicine, Zhejiang University, and was hospitalized. Upon arrival, the patient's body temperature was 36.7 C, pulse rate was 95 beats/min, blood pressure was 138/97 mmHg, respiratory rate was 23 breaths/min, and finger pulse SpO2 was 98%. He was suffering from headache but his neurological signs were normal. The laboratory results are as follows: white blood cell count 7.0 × 109/L, with a neutrophil ratio of 42%, hemoglobin 150 g/L, and platelet count 294 × 109/L; C-reactive protein (CRP)1.8 mg/L; CD4 count 171 cells/μL; normal blood biochemical; negative 1,3-beta-D-glucan test (G test) and galactomannan test (GM test) results (0.1 μg/L). The IgG antibodies of Epstein-Barr virus (EBV) and Human Cytomegalovirus (CMV) were positive, while the IgM antibodies and the test of the nucleic acid of EBV and CMV were negative. The test result for mycobacterium tuberculosis (MTB) was negative based on the blood test with T-SPOT, and the result for cryptococcus capsular antigen test of the blood was positive. The chest CT scan and echocardiography results were normal. The lumber puncture was performed on day 2 and the opening pressure was 285 (cm H2O). The test result of CSF is shown in . The cryptococcus capsular antigen test of CSF was positive. An India Ink microscopy on the CSF sample for cryptococcus was also performed and the result was negative. The results of the blood and CSF smears of bacteria and fungi were negative. The results of the acid-fast staining of CSF smear and GeneXpert were both negative for MTB. The test of the nucleic acid of CMV and EBV in CSF was negative. Based on the negative gram staining and culture study results of CSF provided by the local hospital and the above results, the CSF sample was sent to the laboratory for conventional tests and NGS test (using the Illumina Platform, IngeniGen. Ltd, Zhejiang, China) for pathogen-induced meningitis verification. Contrast-enhanced and diffusion-weighted MRI (3.0T) was performed on day 3, and the partial cerebellar surface enhancement was revealed (), which matched the manifestation of cryptococcal meningitis, and no abnormal signals were detected in the cerebral parenchyma. Two days later, the NGS results showed that streptococcus suis (6 reads) and streptococcus mitis (1 reads) were found in CSF. The results of the blood and CSF culture of bacteria and fungi were still negative on day 5. So, we inquired the patient's medical history again, and he told us he had had close contact with a truck transporting pigs and had choked on water in a water park 2 weeks before his admission. Based on the test reports and his contact history, the diagnosis of streptococcus suis infection was made and intravenous Ceftriaxone (2.0, QD) was given. The patient's headache problem was resolved gradually. A second lumber puncture performed on day 12 revealed improved conditions of CSF (see ), with negative gram staining and culture study results. Ceftriaxone was given to the patient for another 2 weeks and then oral moxifloxacin (0.4, QD) for 2 days. The third lumber puncture was performed on day 17, showing continuous conditional improvement of CSF. The patient was discharged in stable condition on day 21 after 1 week of receiving moxifloxacin. Three weeks later, we performed the fourth lumber puncture to confirm that the meningitis infection was cleared, but the patient's OP level was still abnormal, so fluconazole was given continuously until the OP level and cryptococcus capsular antigen test turned normal 6 months later.


We present the case of a 41-year-old man who presented to the emergency department complaining of swelling in his right groin for two weeks’ duration. He reported that the swelling became painful and non-reducible for the last two days. The pain was associated with decreased appetite and subjective fever. The patient reported having nausea and recurrent episodes of vomiting in the last 24 hours. The patient did not undergo any previous surgeries. His past medical history was remarkable for well-controlled asthma. He was a non-smoker and never drinks alcohol. The patient worked as a school teacher. He has a significant family history of sickle cell disease.
Upon examination, the patient appeared in distress due to the pain. His vital signs were as follows: a temperature of 38°C, a pulse rate of 110 beats per minute, blood pressure of 118/95 mmHg, and respiratory rate of 14 breaths per minute. There was a swelling in the right inguinal region, measuring 4 x 6 cm. It was oval in shape and had a smooth surface with an evident expansile cough impulse. The swelling was non-reducible with slightly erythematous and warm overlying skin.
In view of the clinical features, the diagnosis of incarcerated inguinal hernia was established. The patient underwent a pre-operative laboratory investigation, which revealed leukocytosis with neutrophilia and left shift, elevated erythrocyte sedimentation rate and C-reactive protein, and normal renal and hepatic profiles (Table ). The patient underwent an abdominal computed tomography (CT) scan to identify the content of the hernia and diagnose any possible complications. CT demonstrated a right-sided inguinal hernia that contained the cecum and a rim-enhancing structure that represented an inflamed appendix with surrounding inflammatory changes (Figure ). Such radiological findings conferred the diagnosis of Amyand hernia with acute appendicitis.
The patient was prepared for an emergency laparoscopy operation for the reduction of the inguinal hernia. The patient was administered general anesthesia and was placed in a supine position. Pneumoperitoneum was established. The diagnostic evaluation revealed a right inguinal hernia containing an inflamed appendix. An appendectomy was performed, and the appendix was removed in a sterile bag. The hernia was repaired using the totally extraperitoneal approach with mesh repair. No complications were encountered during the operation. The estimated blood loss was 5 mL and the total operative time was 120 minutes. The patient had an uneventful recovery and was discharged on the fifth postoperative day. The histopathological examination of the appendix confirmed the diagnosis of acute appendicitis.


A previously independent 83-year-old lady presented with a 10-day history of acute confusion, decreased mobility, urinary retention, and constipation. Significant past medical history included ischaemic stroke, atrial fibrillation (AF) on apixaban, polymyalgia rheumatic with bilateral shoulder pain, and obstructive sleep apnoea. The patient was receiving oral acyclovir prior to admission for shingles affecting the genital area. On examination the patient was hypertensive 202/102 mmHg. Extensive bruising was noted over her forearms attributed to anticoagulation. Several tenders, crusted lesions were noted on the labia, with smaller lesions affecting the buttocks and perineum attributed to shingles. Her family reported these were improving with acyclovir. Her cardiorespiratory examination was normal, her abdomen was distended, with urinary retention of 900 mls. Neurological examination showed normal tone, power, sensation and reflexes in all four limbs, and no cranial nerve abnormalities.
Investigations
Blood tests revealed hyponatraemia (128 mmol/L), hypokalaemia (3.2 mmol/L), C-reactive protein (CRP) (23 mg/L), and low platelet count (88 × 109/L). Paired serum and plasma osmolalities were indicative of Syndrome of Inappropriate Antidiuretic Hormone Secretion (SIADH). Initial CT head showed no acute intracranial phenomena. Genital swab confirmed varicella-zoster virus (VZV) infection. Her confusion was initially felt to be multifactorial due to hyponatraemia, constipation, and urinary retention. The patient was catheterised and placed on fluid restriction, anticoagulation was held whilst monitoring platelet count, and given her raised inflammatory markers and genital swab positive for VZV, she was commenced on intravenous acyclovir. Sodium levels improved, however, platelet count and haemoglobin levels declined with no obvious source of bleeding. She exhibited fluctuating cognition and later developed right leg weakness and brisk deep tendon reflexes, which raised the possibility of VZV encephalitis. Lumbar puncture (LP) revealed frankly haemorrhagic cerebrospinal fluid (CSF) (Figure ).
Repeat CT head showed no evidence of intracranial haemorrhage. MRI brain was normal. The patient was commenced on ceftriaxone in addition to acyclovir, whilst awaiting CSF culture results. CSF and radiological findings were discussed with the local tertiary neurosurgical centre, who advised repeat lumbar puncture to exclude a traumatic tap. This again yielded haemorrhagic CSF. Samples were negative for any viral or bacterial pathogens (Table ), and ceftriaxone and acyclovir stopped after 14 days.
The patient exhibited progressive neurological signs in the right lower limb, with increased tone, weakness and upgoing plantar response, and noted new right shoulder pain. She underwent multiple reviews by neurology and discussion with microbiology and the initial concern following her lumbar puncture was regarding possible tuberculosis meningitis or possible, varicella-zoster virus (VZV) encephalitis resulting in bleeding or VZV vasculitis. Further review by the neurology team, who advised a CT aortic angiogram to assess for possible spinal intrathecal haemorrhage. This was reviewed in a neuroradiology meeting and showed blood within the subdural and epidural space of the thoracic spine. MRI spine showed normal CSF appearances above the C4 vertebra (Figures , ) but extensive extra-axial haemorrhage below this extending to the sacrum (Figures , ), with a focal point of high intensity at T1 (Figure ). There was a serpinginous lesion within the lower cervical cord exiting to the right side, suggestive of a lower cervical arteriovenous malformation (AVM) (Figure ). There was also cord oedema with lateral recess narrowing at L4/L5 and L5/S1 with compression of the traversing L5 nerve roots and right S1 nerve roots, in keeping with her lower limb neurology and urinary retention (Figure ).
Treatment
The patient was transferred to a tertiary neurosurgical centre. Repeat MRI spine and magnetic resonance angiography (MRA) with contrast showed a fixed subarachnoid haematoma throughout the spine, but no abnormal vasculature. These findings remained in keeping with an AVM, but at multidisciplinary team discussion, it was felt that neurosurgical intervention would be inappropriate due to extent of spinal cord lesion and oedema present. The patient was repatriated for conservative management with dexamethasone for spinal cord oedema, physiotherapy, and occupational therapy.
Outcome and follow-up
After repatriation, this patient had a long hospital stay whilst receiving physiotherapy and occupational therapy. However, it was not felt that she would return to her pre-admission mobility. At discharge, she continued to have fluctuating cognition, with right lower limb weakness due to spinal pathology and left lower limb weakness due to lack of use. She was discharged to her own home requiring hoisted transfers and a package of care, with a plan for repeat imaging and follow-up at the tertiary neurosurgical centre. This patient died three months after discharge, and it is unclear whether repeat imaging took place.