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A 59-year-old gentleman with end-stage kidney disease due to hepatitis C with focal segmental glomerulosclerosis, on hemodialysis for seven years, underwent deceased donor renal transplantation. The donor kidney had a KDPI of 60%. There was a 4 antigen HLA mismatch with Class II panel reactive assay (PRA) of 62.31%. Class I PRA was 0%. His donor specific antibody testing was positive for an antibody to DQ7. The donor had died as a result of anoxic brain injury following cardiopulmonary arrest resulting from anaphylactic shock. The cold ischemic time was 12 hours, 7 minutes and the warm ischemic time was 51 minutes. Basilixumab was used for immunosuppression induction and the patient underwent early steroid withdrawal. His maintenance immunosuppression was tacrolimus (targeting trough levels 5-8) and mycophenolate sodium 720 mg BID.\nThe patient's postoperative course was complicated by delayed graft function, necessitating continuation of renal replacement therapy. He was hemodynamically stable throughout his hospital course. A renal allograft ultrasound showed good blood flow to the entire kidney with a resistive index of 0.64. A repeat flow crossmatch was negative, but he remained positive for a persistent low-level donor specific antibody to DQ7 (1000 MFI). He underwent a renal transplant biopsy on his 4th day postoperatively. This showed extensive acute tubular necrosis with associated peritubular capillaritis and interstitial nephritis (). Oxalate crystals were seen in several tubules. One large caliber artery showed active endothelialitis, but no tubulitis or glomerulitis seen. C4d staining was negative in the peritubular capillaries. Electron microscopy revealed minimal effacement of podocyte foot processes. The patient received methylprednisolone 500 mg x 3 doses to treat moderate acute cellular rejection. His tacrolimus dose was also optimized as his trough levels had been running low at between 3.5 and 6. He continued to take mycophenolate sodium at a dose of 720 mg BID. A decision was made not to treat for antibody-mediated rejection given that no glomerulitis was seen and that there was minimal capillaritis with a negative C4d stain.\nHe remained dialysis dependent with minimal urine output for three weeks after transplantation. A urine protein : creatinine ratio was elevated to 1070 mg/g when assessed after hospital discharge when his urine output started to gradually improve. His urine microalbumin : creatinine ratio was 450 mg/g. A decision was ultimately made to readmit the patient on day 12 after transplantation for IV thymoglobulin 1.5 mg/kg to treat his Banff Type IIa T cell mediated rejection, given his lack of response to pulsed IV steroid treatment. Given lack of improvement in renal function and urine output by day 16 after transplantation, a second renal allograft biopsy was performed. This revealed residual acute tubular necrosis with associated mild peritubular capillaritis and interstitial nephritis (). Extensive oxalate crystals were now visible in many tubules. His plasma oxalate level was concurrently elevated to 19.3 μmol/L (reference range ≤ 1.9 μmol/L). No signs of persistent acute antibody- or cell-mediated rejection were seen. Again, there was minimal segmental effacement of the podocyte foot processes seen on electron microscopy and no evidence of recurrent focal segmental glomerulosclerosis present.\nHe commenced calcium citrate along with dietary oxalate restriction to manage his hyperoxaluria and his serum creatinine improved to a nadir of 1 mg/dl (88 μmol/L), with a concomitant gradual reduction in his proteinuria and microalbuminuria to undetectable levels. Of note, the patient had no history of malabsorptive intestinal disease and denied any GI symptoms throughout this time period. He had never suffered from renal calculi. At three weeks after transplantation, his urine output and creatinine clearance had recovered sufficiently to enable him to become dialysis independent. A decision was made to continue low-dose oral steroids in the long-term given the presence of early acute cellular rejection on his first allograft biopsy. An interval renal biopsy performed 2 months later showed no ongoing evidence of oxalate deposition, tubular necrosis or cellular rejection. A concurrent repeat plasma oxalate level showed interval reduction to the normal range. |
A 76-year-old man presented with a 5-year history of low back pain. During this period, he had been repeatedly admitted to and discharged from a nearby hospital for lumbar pain. However, he had experienced particularly severe pain for 1 month without trauma and had difficulty walking and lying down due to pain in both lower extremities, and he was referred to our hospital. He had a history of Parkinson's disease and was receiving drug therapy in the Department of Neurology. He had a Hoehn-Yahr classification of III.\nAt his first visit to our hospital, the range of motion of the trunk was severely restricted due to pain. A femoral nerve stretch test was positive and hypesthesia was noted in the bilateral L3 region. Laboratory studies showed that the white blood cell count, erythrocyte sedimentation rate, and C-reactive protein level were normal and that HLA-B27 was negative without findings of sacroiliac arthritis. Radiographs of the thoracic and lumbar spine showed flowing ossification along the anterior and lateral aspects of the Th2-L5 vertebral bodies, with disruption of ossific ridging and marked destructive changes at the L2-L3 intervertebral disc level (). Myelography showed stenosis at the L2-L3 intervertebral space and flexion-extension radiograph indicated instability at this position ().\nFrom these findings, we concluded that the unstable L2-L3 intervertebral space was spontaneous pseudoarthrosis occurring in the presence of DISH and that the motion at the site of pseudoarthrosis was the cause of the severe low back pain and thigh pain. We performed posterior fusion from the L1 to L5 levels with spinal instrumentation, in addition to posterior lumbar interbody fusion at the L2-L3 intervertebral space (). The low back pain and thigh pain during motion disappeared immediately after surgery.\nOne week after surgery, the patient began walking exercises using a body trunk corset, but low back pain and thigh pain redeveloped in week 4. Radiograph findings suggested loosening and back-out of the pedicle screw inserted in the L1-L2 vertebral body. Fixation with a body trunk plaster cast was performed, but there was no improvement in symptoms and abnormal motion was apparent in the L2-L3 intervertebral space. Thus, a second surgery was performed 12 weeks after the initial surgery (). The loosened pedicle screw at the L1-L2 vertebral body was removed and a new screw was inserted into the Th10, 11 and 12 vertebral body. Sublaminar taping was performed for each intervertebral space using polyethylene tape. Posterolateral fusion (PLF) was additionally performed for the L2-L3 intervertebral space using autologous iliac bone.\nAfter the second surgery, low back pain and thigh pain were immediately improved. The second postoperative course was uneventful. The patient became able to walk and to lie down without pain. Plain radiographs taken 1.5 years after surgery showed rigid bony union at the L2-L3 intervertebral space (). |
A 42-year-old woman delivered her first child in 1994 at the age of 25 years. She had prolonged labor, and delivery was complicated by retained placenta; she went into shock after profuse vaginal bleeding and received two blood transfusions at a city hospital. The child died after the 5th day due to birth asphyxia. She had lactation failure and did not resume menstrual cycles after lactation amenorrhea. She also had cold intolerance, constipation and fatigue. She was investigated in July 1996. Investigations revealed central hypothyroidism, growth hormone (GH), PRL and cortisol deficiency on insulin tolerance test []. She was put on treatment in the form of prednisolone 7.5 mg/day, thyroxine 0.75 mg/day and cyclic estrogen/progesterone combination. In October 1996, she complained of polyuria and polydipsia and was investigated for possible diabetes insipidus, which was ruled out on water deprivation test. In October 2002, she noticed three episodes of spontaneous bleeding and was again evaluated. In addition to estimation of basal anterior pituitary hormones, she was subjected to insulin tolerance test to assess GH, cortisol, and PRL reserve; metoclopramide test to assess PRL reserve and gonadotropin-releasing hormone (GnRH) test for gonadotroph reserve. These tests again confirmed GH, cortisol and PRL deficiency. The method and results of these dynamic tests are given below. While she was stable on replacement of thyroxine, prednisolone and intermittent low-dose estrogen/progesterone combination, she came to the hospital with pregnancy of 12 weeks’ duration, confirmed on ultrasound. She was continued on thyroxine 0.1 mg/day and prednisolone 7.5 mg/day. In addition, she was put on progesterone support for 3 months. She delivered in December 2003, had a mild postpartum hemorrhage and received one blood transfusion. After the present delivery, she lactated normally. She resumed infrequent cycles after the lactation amenorrhea. In 2007, incidentally she was again found to have pregnancy of 20 weeks while going for a routine ultrasound; she delivered after a cesarean section and again lactated normally. She continues to have normal lactation at present. She was recently evaluated to test for PRL function again. Metoclopramide test revealed normal stimulable PRL []. Magnetic resonance imaging (MRI) of pituitary revealed evidence of empty sella []. |
We present a case of a 16-year-old girl with severe AN coexisting with juvenile SLE.
The patient was a full-term newborn with normal birth weight. The girl was diagnosed with intrauterine infection which was treated with antibiotic therapy in the department of neonatology. During the first year of life, problems related to weight gain were observed. At the 11th month of age, the girl’s weight was only 8,200 γ (< 3rd percentile). However, coeliac disease was ruled out. Small bowel biopsy confirmed massive eosinophilic infiltration. The patient was diagnosed with food allergy. Due to frequent hospitalisations, infant formula was immediately introduced (as a breast milk substitute) after the birth. The patient’s early childhood development was normal except for speech delay.
The first alarming symptoms of an eating disorder occurred in May 2014 when the girl restricted food intake. Two years later the patient was finally diagnosed with concomitant SLE, the symptoms of which had been previously reported. The sequence of symptoms combined with the patient’s body mass index (BMI) is given in . The subject almost completely refused to eat and drink at the end of 2014 and presented with behavioural disorders (vulgarity, impulsiveness, disinhibition), self-injurious behaviour and suicidal thoughts. In June 2015, the patient’s body weight decreased from 46 to 36 kg (< 3rd percentile) while her height was 155 cm.
The patient was admitted to the department of gastroenterology for the treatment of the eating disorder. Inflammatory changes in gastric and duodenal mucosa were confirmed by endoscopic biopsy. Due to the severe eating disorder, the patient required a high-calorie diet administered by a feeding tube. The diagnosis of AN was established by a consultant psychiatrist and the patient was admitted to the department of psychiatry for further treatment.
Physical examination revealed decreased subcutaneous tissue and muscle atrophy. The weight of the patient was 35.3 kg (BMI 14.5 kg/m2 |
A 41-year-old woman presented with abnormal vaginal bleeding for 2 months. She had a history of two cesarean deliveries and one spontaneous abortion. She had undergone excision of a uterine adenomyosis 3 years previous. Her latest pregnancy resulted in the delivery of a full-term baby 1 year previous. The physical examination was otherwise normal. Pelvic ultrasonography showed focal thickening of the endometrium. She received endometrial curettage with a clinical diagnosis of endometrial hyperplasia. A serum β-human chorionic gonadotropin (β-hCG) measurement was performed 1 week later because an IT tumor was suspected after pathologic examination of the endometrial curettage specimen. The β-hCG level was not elevated (1 mIU/ml; reference range: 0 ~ 5 mIU/ml). The abdominal and pelvic computed tomographic scans exhibited an enlarged uterus with an irregular contour and heterogeneous contrast enhancement without para-aortic or pelvic lymphadenopathy. No metastatic lesion was detected on a chest x-ray. She underwent a total abdominal hysterectomy. Neither postoperative chemotherapy nor radiotherapy was given. She was alive with no evidence of local recurrence or distant metastasis 30 months after the hysterectomy.\nOn macroscopic examination, the uterus measured 9.0 × 8.3 × 4.2 cm and was slightly enlarged. The serosa showed focal fibrosis. The previous CS site of the lower uterine segment revealed a markedly thinned myometrium. There was an irregular, plaque-like, soft, brownish tumor centered at the CS site (Figure A, B). The tumor was 3.0 × 2.0 cm in dimensions and 0.9 cm in thickness. It had invaded the myometrium but was still confined to the uterus.\nHistologically, the tumor consisted of variably sized nodules with central fibrinoid necrosis in the endometrium and myometrium of the lower uterine segment (Figure A). Suture material and suture tracts due to the previous CS were observed. The nodules commonly revealed an infiltrating border in the myometrium. The center of the nodules was composed of mononucleate epithelioid cells arranged in cohesive sheets (Figure C, G). The background contained hyaline extracellular matrix. The tumor cells had distinct cell borders, clear cytoplasm, and relatively uniform round nuclei with fine chromatin. Enlarged hyperchromatic nuclei were occasionally found. Mitotic figures were infrequent. Epithelioid clear cells revealed diffuse and strongly positive immunoreactivity to pan-cytokeratin (clone AE1/AE3, 1: 400; Dako, Carpentaria, CA, USA), p63 (clone 4A4, 1: 100; Santa Cruz Biotechnology, Santa Cruz, CA, USA), and HLA-G (a gift of Dr. Ie-Ming Shih) (Figure D). Human placental lactogen (hPL) (1: 250; Pierce Biotechnology, Rockford, IL, USA) and CD146 (1: 50; Pierce Biotechnology) were negative (Figure E, F). No cell immunoreactive with hCG (1: 300; Dako) was found. Ki-67 (clone MIB-1, 1:50; Dako) labeled 12% of tumor cells. The morphologic and immunohistochemical features of the epithelioid cells were characteristic of an ETT.\nIn the periphery of the nodules, larger pleomorphic cells, which histologically and immunohistochemically differed from tumor cells of the ETT, were seen (Figure C, H). They were polygonal with abundant eosinophilic cytoplasm and relatively obvious nuclear atypia. They had invaded the myometrium in cell cords, dissecting and separating the smooth muscle bundles. They were immunoreactive for CD146 and hPL but negative for p63 (Figure D-F). Ki-67 labeled about 10% of the tumor cells. These histologic and immunohistochemical findings were consistent with features of a PSTT. At the interface of the ETT and PSTT components, both type of tumor cells merged imperceptibly.\nIn addition, several microscopic hypocellular nodular lesions, closely associated with the mixed ETT and PSTT, were found in the endometrium of the lower uterine segment, the surface of the endocervix, and the fibrous scar of the CS (Figure B). They had circumscribed margins and were round or plaque-like in shape. They were composed of mononucleate ovoid cells arranged in single cells and small sheets in a hyaline background. These cells had uniform nuclei with fine chromatin. The immunohistochemical findings were the same to those of the ETT. Compared to the ETT, the nodular lesions were smaller in size and paucicellular. No necrosis was found. The Ki-67 proliferation index was <5%. Based on the histologic and immunohistochemical features, the smaller nodular lesions represented PSNs. In summary, the tumor represented a rare example of a coexisting ETT and PSTT that had likely transformed from PSNs. Microscopic findings of the endometrial curettage specimen were similar to those of the hysterectomy specimen, except that no PSNs were present in the former. |
A 62-year-old lady sought an ophthalmology consultation for complaints of drooping of right eyelid since 10 days. She was referred to our endocrinology clinic thereafter. The drooping of the right eyelid was sudden and was better at the start of the day and got worse as the day progressed. When she lifted the lid with her hands, she also noted double vision. On questioning, she had complaints of watering from both eyes and occasional redness with a gritty sensation since a year. Her daughter notes that her eyes have become more prominent since the last year. She had no past history or family history of thyroid illness and had no complaints suggestive of thyrotoxicosis although she had constipation and malaise. She had no difficulty in swallowing food or speaking. She had no difficulty in getting up from the squatting position, or climbing stairs or combing her hair. She had no history of cough, hemoptysis or shortness of breath.\nOn examination, she was a moderately built lady, conscious oriented and alert. General examination revealed a visible diffuse goiter, dry skin with delayed relaxation of ankle jerks. Examination of the eyes revealed bilateral proptosis (26 mm in the left eye and 24 mm in the right eye). The right eye revealed ptosis with transient improvement after rest []. There was no exotropia and esotropia. Ocular movements testing revealed restriction of upward gaze and adduction both eyes. Pupils were equal and reactive to light. Clinical activity score was 1/7 in both eyes (right – conjunctival congestion and left pain at rest). Vision was normal in both eyes. Examination of the central nervous system revealed no bulbar weakness or weakness in limbs. Sensory system examination was normal.\nA provisional diagnosis of TAO, hypothyroidism and myasthenia gravis was made. Routine investigations were normal. Thyroid function tests revealed primary hypothyroidism (thyroid-stimulating hormone 38 mIU/L, Free T4 0.8 ng/dl, anti-thyroid peroxidase 189 IU/L). Computed tomography of the orbit [] showed bilateral proptosis (left > right) with no mass lesions in the orbit. shows enlargement of extra ocular muscles both eyes, especially inferior recti and medial recti, findings characteristic of TAO.\nA neurology consultation was obtained. Anti-acetylcholine receptor antibody (antiAchR) was positive 2.32 nmol/L (N <0.5 nmol/L). A repetitive nerve stimulation test at the limbs was normal but the testing at the nasalis muscle was found to inconsistently positive. A diagnosis of ocular predominant myasthenia gravis was made. A computerized tomography thorax was normal. Magnetic resonance imaging of the brain was normal.\nShe was started on Levothyroxine 75 mcg/day and pyridostigmine 15 mg thrice daily and showed improvement in general well-being and ptosis. |
A 54-year-old male with no significant past medical history was admitted with acute left leg pain for 3 days and acutely worsening chronic cough. He was found to be hypoxic on admission (SpO2 90% on room air) and was placed on oxygen support. He was confirmed SARS-CoV-19 virus PCR test positive. D-dimer level on admission was 6,802 ng/mL, and DUS showed isolated popliteal artery occlusion with reconstitution of tibial arteries as well as soleal vein thrombosis (, panel A). Additionally, he was diagnosed with pulmonary thromboembolism. Therapeutic anticoagulation with heparin was administered. His chest X-ray was abnormal with multiple areas of infiltrates. Given his tenuous pulmonary status, the vascular team deferred emergent lower extremity revascularization. After initial improvement of the patient’s foot pain, the clinical condition worsened with an increase in ischemic pain and deterioration of the neurologic status of the affected limb on day 8. The vascular team decided that an emergent peripheral angiogram should be performed due to imminent limb loss. An intraoperative angiogram demonstrated a second lesion in the left common iliac artery (, panel B) in addition to the earlier popliteal lesion. Surgical intervention was performed with embolectomy of the popliteal and common iliac artery. The completion angiogram revealed excellent blood flow to the left lower extremity. Intravenous heparin was discontinued post-procedure. The postoperative course was unremarkable, and anticoagulation therapy was resumed on the third postoperative day. During surgery, gray-white thrombi were retrieved, and clot pathology demonstrated platelet-rich thrombus with red blood cells and minimal fibrin (). Therefore clopidogrel therapy was initiated. After further discussion with the vascular team, the patient was discharged home on oral anticoagulation (due to PE/DVT) and clopidogrel (for arterial thrombosis) with complete recovery on day 14.\nAs we continue to learn more about the pathologies associated with SARS-CoV-2 infection, it is essential to mention that our document reflects our institutional experience and might be subject to modification in the near future. Additionally, our considerations certainly apply to regions that are considered epicenters of the pandemic. Nevertheless, in view of the currently increasing numbers of infections in many other regions of the world our insights constitute a differentiated approach that may help others to identify the cases in which procedures can be safely postponed in order to avoid an exhaustion of the health care system but also the cases in which a procedure should not be postponed to avoid further harm for the patient. The application of this suggested triage system will facilitate the ongoing care of PAD patients during the current pandemic. |
A 14-year-old South Asian boy from rural Bengal (India), born of a second degree consanguineous marriage, with normal birth and development history, presented with abnormal brief jerky movements involving his trunk and limbs, with recurrent falls for 10 months. The jerks were neither stimulus sensitive nor present during sleep. No loss of consciousness was reported to occur with these jerky movements. Recurrent convulsions involving the left half of his body, without impairment of awareness, was present for 8 months. It was followed by insidious onset of mild weakness of the left half of his body for 7 months. Subsequently he suffered progressive decline in his general ability to maintain average daily activity independently for 5 months. He had to discontinue schooling because of his failing cognitive functions. For 2 months prior to presenting to us, he developed rapid dance-like movements involving all four limbs that flowed from one muscle to the other in a more or less continuous fashion. Occasionally it would become somewhat flinging particularly in his upper limbs. There was no history of similar illness in the family. He received all the scheduled vaccines as was stated by his mother.\nThe height of the boy was 150 cm and he did not have any dysmorphic facial features. A clinical examination revealed generalized choreiform movements as the most obvious finding. These movements intermittently became flinging in nature, resembling ballism. Generalized myoclonic jerks were seen embedded inside the flurry of chorea-ballism. When he was asked to protrude his tongue, besides motor impersistence, oromandibular dystonia was also found. He had severe dysarthria with apparently preserved comprehension. A limited cognitive assessment revealed reduced attention span as well as short-term memory impairment. Rigidity was obvious in all four limbs along with dystonia in both lower limbs. Weakness in the left half of his body along with brisk reflexes and extensor plantar on left side was also detected on motor system evaluation.\nRoutine laboratory parameters revealed impaired fasting glucose (120 mg/dl), mildly raised liver enzymes and creatine phosphokinase (CPK) level of 820 IU/L. Other blood and urine parameters were within normal limits. Screening investigation for Wilson’s disease, storage disorders, and metabolic disorders were all negative. A routine cerebrospinal fluid (CSF) study was unremarkable and anti-measles antibody was negative. Anti-nuclear antibody in blood was also negative. His serum level of lactate was 36 mg/dl (2–19 mg/dl) while CSF lactate was 42 mg/dl. Shortening of PR interval (0.10 second) was found in electrocardiography. Two-dimensional echocardiography was devoid of any abnormality. Serial brain imaging was done at different centers throughout the course of his illness. On studying his MRI brain images sequentially, a relapsing remitting pattern of lesions was detected. On T2/fluid-attenuated inversion recovery sequence (FLAIR) there were hyperintense lesions that mainly involved subcortical white matter in frontoparietal areas (Fig. ). An area of diffusion restriction was found in the right capsule-ganglionic region (Fig. ) that temporally coincided with the onset of left hemiconvulsions and hemiparesis. Magnetic resonance spectroscopy (MRS), done at our center, showed the presence of lactate peak in brain lesions. Brainstem auditory response revealed bilateral prolonged latency. Electromyography (EMG) showed short duration low-amplitude polyphasic motor unit action potential which was suggestive of myopathic pattern. Spike-wave discharges were observed arising from bilateral frontal areas on electroencephalography (Fig. ). A muscle biopsy, which was done from left vastus lateralis, revealed ragged red fibers (Fig. ), suggestive of mitochondrial failure and deposition of abnormal mitochondria below the plasma membrane of muscle fibers.\nAccording to the clinical criteria, MELAS syndrome was the most probable diagnosis in our case and we needed to confirm the diagnosis. As a facility for analysis of respiratory chain enzymes in the muscle was not available, we decided to search for underlying genetic abnormality in mtDNA. A polymerase chain reaction (PCR) method was employed for this purpose. Amplification of DNA in whole blood sample of our patient was performed for detection of mutations 3243A>G, 3271T>C, and 3251A>G in mitochondrial tRNA leucine 1(MT-TL1), by using appropriate wild type and mutant type specific primers for each and a common reverse primer for all. Genetic analysis result was as following: A>G point mutation at position 3251 of MT-TL1 gene of the mtDNA with heteroplasmy of 70%.\nAfter reaching the diagnosis, valproate was taken off and lamotrigine was introduced. He was put on co-enzyme Q supplement and haloperidol for abnormal movements. Six months into follow-up his seizures and abnormal movements were controlled significantly with slight improvement of cognitive abilities. |
A 52-year-old right-handed man, who suffered from hypertension for 5 years without regular medicine and followup, was admitted to our facility after acute loss of consciousness. The patient exhibited left hemiplegia secondary to right putaminal hemorrhage (3 × 5 cm in size) extending into the posterior portion of the internal capsule with the mass effect. On neurological examination, the patient had weak muscle power (1/5 in the left upper limb and 2/5 in the left lower limb) and positive Babinski sign on the left side. An emergent craniotomy for removal of a hematoma was performed; however, the patient demonstrated no significant recovery in his left hemiplegia after the operation. Results of a Romberg test and dynamic testing were poor. The patient's Functional Independence Measurement (FIM) score was 50, and his Barthel index (BI) was 25. Amlodipine besylate 5 mg qd, valsartan 80 mg qd, and atorvastatin 10 mg qd were prescribed for secondary prevention of cerebrovascular disease. Color Doppler sonography showed only mild degree of intimal thickening and sparse mural calcified plaques along the course of bilateral extracranial common and internal carotid and vertebral arteries as well without significant stenosis. MR angiography of the circle of Willis shows decreased signal intensity and number of branches of right middle cerebral artery.\nAfter intensive rehabilitation, the patient's hemiplegia remained, but his gait improved gradually. Two months after the stroke, muscle power was reevaluated in the left upper and lower limbs and was 2/5 and 3/5, respectively. Single photon emission computed tomography (SPECT) showed decreased perfusion in the right cerebral cortex and in the left cerebellum. CCD on the left side was confirmed (). Four months after the stroke, the patient noticed that he involuntarily stretched his hemiplegic arm when yawning in bed. The movement consisted of a progressive abduction, anteroflexion, and mild internal rotation of the shoulder, followed by arm lifting with a flexion of the elbow. The movement lasted for a few seconds. The involuntary activity was absent when the patient was in a sitting position. Repeated attempts to induce yawning by imitation were unsuccessful.\nEight months after the stroke, a follow-up SPECT showed persistent CCD (). The patient's arm was still plegic, but he could walk slowly using a cane with the assistance of an ankle-foot orthosis. The follow-up FIM score and BI were 102 and 70, respectively. The patient continues to experience persistent hemiplegia and CCD, and his functional index has improved only gradually. The frequency and intensity of the phenomenon increased steadily with time after the stroke and also occured when sitting. |
A 64-year-old male presented with a two month history of difficulty urinating and was found to have a fungating penile mass involving 50% of his penis. The mass was hard and fixed and extended from the glans proximally up the shaft. He also had bilateral palpable inguinal lymphadenopathy. There were no associated constitutional symptoms. Given there was a high suspicion for malignancy, the patient underwent partial penectomy within a month of presentation. Biopsy results confirmed a pT2 tumor with invasive keratinizing squamous cell carcinoma, poorly differentiated, and tumor size of 5 × 4 × 2.5 cm, with corpus spongiosum and lymphovascular involvements.\nFollowing the procedure, the patient had PET-CT for staging, and imaging revealed enlarged hypermetabolic bilateral axillary lymph nodes concerning for metastatic disease. In addition, there was a large centrally necrotic lymph node conglomerate in his left groin that had increased FDG avidity. The patient had left inguinal and bilateral pelvic lymph node dissections revealing metastatic squamous cell carcinoma in multiple lymph nodes. The left inguinal mass was also found to be metastatic well-differentiated SCC. His diagnosis was staged at T2N3M0.\nAfter his surgical procedures, patient was started on adjuvant chemotherapy. He began first line chemotherapy with paclitaxel, ifosfamide, and cisplatin (TIP). He underwent 4 cycles of TIP but eventually developed disease progression on repeat imaging. At this point, the patient was started on cetuximab given EGFR amplification on tumor analysis with the FoundationOne testing platform. However, the patient had an allergic reaction to cetuximab, so his treatment was changed to panitumumab. The patient had stable disease and a progression-free survival of 6 months with anti-EGFR treatment, which is clinically significant given that this treatment was given in the second-line setting for an aggressive tumor type that other than chemotherapy there is no other approved drug to date.\nThe patient was ultimately started on the PD-1 inhibitor nivolumab. He had initial response to immunotherapy followed by stable disease, so he had a disease control rate of an additional 6 months with this investigational agent at that time. Ultimately, he was placed on hospice and passed away two years from the the time of diagnosis. |
A 10-month old girl presented with history of abdominal distension since 45 days associated with fever, cough and weight loss. There was a history of malignancy in multiple members of the maternal side of the family; however the exact details were not available. On examination a right-sided abdominal mass was found. On ultrasonography a 9 cm x 7 cm heterogeneous predominantly echogenic mass with multiple internal anechoic areas along anterolateral aspect of right kidney, causing its posterior displacement was reported. There was another well-defined 4.8 cm x 4.3 cm heterogeneous, predominantly echogenic mass in right suprarenal region causing inferior displacement of the kidney. Contrast enhanced computed tomography (CECT) showed a similar picture of the two masses (Fig. 1). Chest radiograph showed no lesion. Trucut biopsy from the mass showed tubular and papillary structures lined by tall columnar cells, showing nuclear hyperchromasia with atypia and mitosis, suggestive of epithelial component of WT.\nAs per SIOP protocol, a diagnosis of WT (localized disease) was made and the patient was given vincristine and actinomycin-D for four weeks. Repeat CECT did not show significant reduction in tumor size. Surgery was then planned. On exploration, there was a 15 cm x 10 cm x 8 cm mass involving the right kidney, and another 5 cm x 5 cm x 3 cm mass located separately above the right kidney in the region of suprarenal gland (Fig. 2). Enlarged supra-hilar, hilar, infra-hilar and mesocolic lymph nodes were also found. Patient underwent right nephroureterectomy with excision of suprarenal mass and lymph node sampling. There was no tumor spill. In postoperative period patient developed adhesive intestinal obstruction requiring repeat exploration with adhesiolysis on day 5.\nThe final histopathology report showed WT of the right kidney with predominantly epithelial component (resected margins were free of the tumor) (Fig. 3). The right suprarenal mass showed undifferentiated NB involving the capsule (Fig. 4). The hilar and infra-hilar nodes showed tumor deposits of WT. A thorough work-up for risk factors and metastasis of neuroblastoma was done. It included skeletal survey, meta-iodobenzyl guanidine (MIBG) scan, 24-hour urinary vanillyl mandelic acid levels and bone marrow aspiration biopsy. They were within normal limits. However, N-myc amplification in tumor tissue was positive. Thus, a final diagnosis of WT stage III (intermediate risk) with NB stage I (high risk) was made. The WT was of higher stage and was thus targeted first with radiotherapy (21 Gy radiation over 5 weeks) given to tumor bed with opposite kidney shielding. The patient was then put on vincristine (oncovin), cisplatin, etoposide and cyclophosphamide (OPEC chemotherapy) for NB. During the second cycle of chemotherapy she developed metastatic NB in the left supraclavicular lymph nodes. Repeat CECT scan showed marked retroperitoneal lymphadenopathy and MIBG scan showed suspicious increased uptake in left superior mediastinum. Thereafter, the child had a stormy clinical course. She developed left sided hemorrhagic pleural effusion (cytology showed reactive mesothelial cells in hemorrhagic background) with respiratory distress that was not responding to intercostal drain insertion. In view of unresponsiveness to OPEC, she was begun on rapid COJEC (cisplatin, oncovin, carboplatin, etoposideand cyclophosphamide) chemotherapy. However, her clinical condition continued to deteriorate. She developed decreased oral intake as well as axillary lymphadenopathy followed by abdominal distension and finally succumbed to metastasis. |
A 13-year-old male was referred to the pediatrician as atrioventricular conduction abnormalities were coincidentally observed during preoperative screening for dental surgery. His resting electrocardiogram (ECG) showed no other abnormalities. The 24-h Holter monitoring revealed frequent sinoatrial arrests (N = 77, up to 2.75 s) and a second degree atrioventricular conduction block type I and II (). No ventricular events were recorded. He did not have any complaints nor did he ever have a syncope. He participated in school gymnastics without any restraints. Physical examination was normal and echocardiographic examination also revealed no abnormalities. His family history was positive for cardiovascular disease. His grandmother had a myocardial infarction at the age of 75 years and his uncle at the age of 45 years. Another uncle died suddenly at the age of 30 years. Both uncles were brothers of his mother.\nAt the age of 17 years, 24-h Holter monitoring again showed a second degree atrioventricular conduction block type I and sinoatrial arrests (N = 224), but now with increased duration up to 3.48 s. The only ventricular events were four premature ventricular beats. During the following visit to the outpatient clinic, he complained of excessive exhaustion, dizziness and dyspnea during exercise and one episode of syncope. Additional cardiac evaluation included a signal averaged ECG and cardiac imaging. The signal averaged ECG was positive as two of the three parameters are abnormal including a low-amplitude signal duration of 44 ms (normal duration < 38 ms), and a root-mean-square voltage in the last 40 ms of the QRS of 12 µV (normal value: > 20 µV). The MRI showed a dilated, right ventricle (RVEDV/BSA: 133 mL/m2) with an ejection fraction of 41% and dyskinesia of the right ventricular anterior wall (). The ECG revealed inverted T waves in leads V1 and V2 in the presence of an incomplete right bundle-branch block (). Hence, the diagnosis of ARVC could now be made as one major and two minor criteria were fulfilled []. Because of his dysrhythmias, a DDD pacemaker was implanted, after which all his complaints resolved. |
A 27-year-old woman was referred to our center with a painless palpable mass in the upper outer quadrant of the right breast. She had no family history of breast cancer and no personal history of breast injury. A physical examination revealed a solitary, hard, nontender, and nonmobile mass. There were no palpable lymph nodes in the axilla. Mammography showed an approximately 3×2 cm oval-shaped, microlobulated, hyperdense mass in the right upper outer quadrant of the breast. There were some lucent centered and coarse heterogeneous microcalcifications noted within the mass (). Ultrasonography (US) of the breast showed an irregularly shaped, microlobulated, hypoechoic mass with combined posterior features that was approximately 3 cm in diameter. A color Doppler study showed increased vascular flow to the peripheral portion of the lesion (). Magnetic resonance imaging (MRI) revealed an approximately 3×2 cm irregular mass with heterogeneous enhancement and dark internal septations on dynamic contrast-enhanced images. The MRI showed a rapid rate of enhancement in the initial period and washout in the delayed period of enhancement (). A kinetic curve of the lesion showed rapid initial enhancement and a washout pattern. The MRI showed no evidence of metastasis to the internal mammary, supraclavicular, and axillary lymph nodes, or to the mediastinum. We classified the mass as Breast Imaging Reporting and Data System category 4C. An US-guided core biopsy was performed and the pathologic results were consistent with an IMT with atypical features. Surgical excision of the tumor was then performed. Gross examination of the surgical specimen revealed an approximately 3×2 cm mass that was firm and yellow in color. Microscopy showed a well-circumscribed tumor with high cellularity. The tumor predominantly consisted of spindle cells with a swirling storiform-like pattern as well as inflammatory cells including plasma cells and lymphocytes (). Although the mass showed high mitotic activity (10 mitoses per 10 high-powered fields [HPFs]), the mitotic figures and the nuclear features were not significantly atypical (). Furthermore, the mass showed an absence of cells with epithelioid morphologies suggestive of carcinoma in situ or invasive carcinoma. Immunohistochemically, the spindle tumor cells were focally positive for smooth muscle actin (SMA) with a tram-track staining pattern () and were focally positive for desmin, but were negative for cytokeratin (CK), low molecular weight CK, high molecular weight CK, p63, and anaplastic lymphoma kinase (ALK). Approximately 30% of the spindle-shaped tumor cells were positive for Ki-67 (). The resection margins were clear of tumor cells. Collectively, the histopathological and immunohistochemical findings supported the diagnosis of IMT, which resulted in the exclusion of the possibility of spindle cell metaplastic carcinoma. A diagnosis of IMT was made, and the patient was advised to have regular follow-up assessments given the rare cases of local recurrence that had occurred with a time to recurrence that varied between 3 months and 9 years []. Our patient had regular postoperative follow-up every 3-6 months for 1 year with US. However, the patient was lost to follow-up after 1 year.\nOne year later, the patient was admitted to our breast center with newly developing masses in the upper inner quadrant of the right breast and right cervical area. A physical examination revealed a solitary hard, nontender, nonmobile mass that measured 5×4 cm in the upper inner quadrant of the right breast and a 3×1 cm mass in the right cervical region. Ultrasonography of the breast showed an approximately 7 cm, irregularly shaped, hypoechoic mass with a circumscribed margin in the right internal mammary chain and an enlarged, hypoechoic lymph node (approximately 3×1.7 cm) with eccentric cortical thickening in the right supraclavicular area. Chest computed tomography (CT) showed a 7.9×7.9 cm irregularly shaped, heterogeneously enhanced metastasis to the internal mammary chain and two supraclavicular lymph nodes measuring 3.3×1.6 cm and 1.5×1.5 cm on postcontrast enhanced images. The metastasis to the internal the mammary chain was noted to have anterior mediastinum, right pectoralis major muscle, and left innominate vein involvement that was confined to the superior vena cava. Furthermore, bone destruction in the sternum and right first and second ribs was noted on chest CT (). Combined 18F-fluorodeoxy-glucose positron emission tomography (PET) and CT showed metastasis to the internal mammary chain, supraclavicular lymph nodes, and mediastinal lymph nodes (). An US-guided core biopsy was performed, which showed that the tumor was mainly composed of proliferating spindle cells arranged in a loose fashion and surrounded by infiltrating inflammatory cells (). There was increased cellularity and spindle cell atypia compared to the findings from the first excisional biopsy. The tumor cells showed high mitotic activity (10 mitoses per 10 HPFs) (). In addition, partial adjacent muscular invasion was noted. Immunohistological studies revealed that the tumor exhibited positive staining for desmin, SMA (), and CK, but was negative for S-100, ALK, and p63. Positive Ki-67 staining was observed in 40% of the cells (). These clinical and pathological findings confirmed the diagnosis of metastatic IMT with malignant transformation. |
A 69-year-old male patient presented to a secondary hospital emergency department with a sudden headache followed by momentary loss of conscience and cranial nerve paresis. After 18 days of the initial symptoms, he was transferred to our hospital. By physical examination after admission, the patient presented with slight headache and neck stiffness, blood pressure on left arm of 140 mmHg × 110 mmHg and right arm of 170 mmHg × 100 mmHg. Neurological exam showed a conscious and oriented patient with right oculomotor (III) and trochlear (IV) paresis. The CT scan revealed a Fisher Grade III SAH []. The subsequent four-vessel DSA revealed a right true PCoA aneurysm [Figure and ], occlusion of the left subclavian artery [], right vertebral artery steel phenomena to the left vertebral artery [] and >80% right ICA stenosis. The therapeutic discussion was based on the fact that the steel was so important that no enhancement of the superior segment of the basilar artery and its rami were seen on posterior circulation angiogram, and were mainly supplied by the right PCoA. In this manner we believed that the aneurysm might be related to the high flow through the PCoA, despite the important stenosis in right ICA. Our hypothesis was that normal flow restoration due to correction of the left subclavian artery obstruction would correct the steel phenomenon, diminishing the high flow through the PCoA, with possible aneurysmal exclusion. The patient underwent recanalization and stent placement on the left subclavian artery [], resulting in restoration of flow through the left vertebral artery []. Full replenishment of the vertebra-basilar circulation and posterior cerebral arteries (PCAs) [] was observed. The flow through the PCoA was lowered and contrast stagnation inside the aneurysm was observed []. Late control angiogram of the aortic arch and right ICA showing adequate flow through the left subclavian artery and aneurysm's circulation exclusion []. |
A 68-year-old male, ex-serviceman, presented with a history of open prostatectomy (Millins). The procedure was operated elsewhere and presented with a complaint of a bulge in the abdomen along a previous scar site. He did not have any complaints of altered bowel and bladder habits. He did not have any other specific complaints. On evaluation, he gave history of surgery 1 year back. One year ago, he underwent Millins procedure for benign prostatic enlargement (BPE). This procedure involves removing part of the prostate using a transcapsular retro pubic approach (extra peritoneal) through a cut in the abdomen. Two weeks after the surgery, he developed abdominal wound dehiscence. Once the wound granulated well, he underwent secondary suturing of the abdominal wound after 2 months of surgery. He developed incisional hernia 3 months later. He continued to have protuberant abdomen with visible bowel peristalsis. He was reassured and advised to wear abdominal binder. On examination, his general condition was good. There was a bulge of size ~25 × 10 cm projecting from his anterior abdominal wall at the site of the previous scar ().\nA palpable midline rectus defect of 10 cm was noted. Visible bowel peristalsis was seen. Other system examinations were normal. Routine laboratory investigations were normal, and he did not have any comorbid illness. Computed tomography (CT) of the abdomen confirmed thinning of the rectus sheath with focal outpouching of rectus in the infra-umbilical region and herniation of small bowel loops with loss of domain (LOD) ().\nHe was planned for surgical exploration. On exploration, a defect in midline for ~8 cm in width was noted. Rectus was retracted laterally and could not be brought easily to the midline. Adhesiolysis was done. It was decided to go ahead with posterior component separation with TAR since the defect was very wide. The procedure commenced with the separation of posterior rectus sheath from the anterior rectus at ~1 cm from the midline where the previous linea alba was present. Retro rectus dissection was done till the level of linea semilunaris. Care was taken to preserve the neurovascular bundles encountered. Incision was made on internal oblique fascia and the transverse abdominis muscle was hooked and divided using an electrocautery. The transverse abdominis muscle fibers were released along its entire insertion line at the level of semilunaris extending from xiphoid process above. Inferiorly, it was separated till the level of arcuate semilunaris below which the muscles were deficient and only peritoneum was present. Laterally, the release process was extended till bilateral psoas muscles were visualized. Superiorly, it was extended till the central tendon of diaphragm. The posterior rectus sheath was approximated in midline using non-absorbable sutures after placement of intra-peritoneal drains. Polypropylene mesh of size ~30 × 15 cm was placed over the posterior rectus sheath covering in a sublay fashion and was secured. Suction drain tubes were placed over the mesh covering and the anterior rectus sheath was approximated in the midline without tension. Skin closed in the midline (). Daily vitals and drain output was monitored. After considerable decrease in drain output, it was removed on the fourth post-operative day. Abdominal sutures were removed at the end of second post-operative week. He was subsequently discharged a week later and was put on abdominal binder. Patient attended his routine outpatient visit after 2 weeks with no complaints. |
A 44-year-old male patient visited the hospital. He claimed low back pain at right, buttock pain, and thigh pain at upper front with fever that occurred 6 days ago. He had got physical therapy and medicinal herbs treatment for a long time in the oriental medical clinic because of low back pain. In recent, he got additional pharmacopuncture treatment several times until 6 days before visiting this hospital. Right after the pharmacopuncture treatment, the symptom was improved, but he got a fever and the degree of pain got worse for 6 days before visiting this hospital. At first in this hospital, the patient claimed that he felt stiff in the lumbar at right side, buttocks, and thigh at upper front and he said they were numb. The degree of pain was 6/10 on visual analogue scale (VAS). The pain was sustained all day without being relieved even though taking a rest. It got worse when bending over or straightening his back, and it was greatly serious at night, so he claimed sleep disorders. The specific medical history of the patient, which can cause infection disease was not identified.\nIn the physical examination, tenderness was detected in spinous process of lumbar spine at level 2, 3, 4, and the right paraspinal area. There was no particular opinion in other neurologic examination or physical examination. High fever of 39.7 degrees was shown but other vital signs such as blood pressure and pulse were normal. In blood test, white blood cell count was 7,300/µl, which is in normal range, and erythrocyte sedimentation rate and C-reactive protein were respectively 67 mm/hr, 97.44 mg/L. AST/ALT/γGT were 92/151/226 IU/L, and ALP increased to 152 IU/L.\nThere was no particular opinion in simple radiography of lumbar spine, but in MRI for checking lesion, a wide range of myositis and multiple abscess in paraspinal muscles and psoas muscle between right lumbar spine at level 3-5, including multifidus muscle, longissimus muscle, iliocostal muscle, quadratus lumborum muscle, psoas muscle were detected (). With blood culture, about 3 cc thick pus was aspirated () from three abscess sites () under ultrasound needle guide and it was cultured. Except for it, there was no particular opinion in simple radiography of chest, ECG, echocardiography, and urine test.\nBefore obtaining the results from culture, 3rd generation antibiotic cefazolin and gentamicin were injected under being admission. As a result of incubation, staphylococcus aureus was identified, and cefazolin and gentamicin treatment which sensitivity checked were maintained. From the 4th day after admission, the fever came down significantly, the symptom was improved, and the patient's condition was stabilized. The patient discharged after replacing with levofloxacin which is the oral antibiotic 12 days after admission. Since then, the patient's condition was improved, but the treatment sustained for 1-month more with the oral antibiotic and it was closed. |
On November 2009 a 33-year old woman with no significant previous medical history was referred to her gynecologist due to complaints of pelvic pain. A right ovarian cyst was diagnosed upon examination. However, due to persistent pain, a CT scan was performed that revealed a right pelvic mass. On 12 November 2009 the patient’s gynecologist performed laparoscopic surgery during which a sub peritoneal lesion likely to start from the bladder was found. The mass was removed but ruptured during surgery, with intraoperative spillage of mucinous material. A cystoscopy was performed postoperatively, which showed a reddish lesion of the dome of the bladder. The intraoperative histological diagnosis was mucinous adenocarcinoma. This was then confirmed by the final histological examination.\nThe patient was referred to our hospital, Istituto Nazionale Tumori (National Tumors Institute), Milan, Italy, a referral center for the treatment of oncological disease in Italy, and a histological review was performed by our genitourinary pathology expert. The immunohistochemical analysis was positive for CDX-2 and CK20 and negative for CK 7, suggesting a diagnosis of mucinous adenocarcinoma originating from the urachal ligament. We then performed a whole body CT scan that showed two metastases in the right lung, one at the lower lobe and one in the middle lobe, with a diameter of 15.6 and 8.5 mm, respectively, and one lesion anterior to the bladder wall and the dome that extended through the bladder wall, protruding into the lumen. Serum CEA was 15.39 ng/mL (normal <5), CA 19.9 was 70.1 U/mL (normal <37), CA 125 negative, CA 15.3 negative.\nWe discussed the case of the patient with the urological surgeon and, given the extension of the disease, we decided to treat the patient with systemic chemotherapy rather than performing surgery.\nThe histological type, the strong mucinous component and the phenotypic similarities with a cancer of gastroenteric origin, rather than urothelial, prompted us to use the association of three drugs: irinotecan 180 mg/m2 (300 mg tot.) on day 1, oxaliplatin 85 mg/m2 (145 mg tot.) on day 2 and capecitabine 2000 mg/m2/day (days 2-6); cycles were repeated every 2 weeks. This association was undertaken based on the clinical experience of our group in gastrointestinal tumors.\nWe started this chemotherapy regimen on 16 Dec 2009 and continued for 6 cycles, until 03 March 2010, with evidence of radiologically stable disease on CT scans after 3 and 6 cycles, and biochemical response (CA 19.9: 15.2 U/mL, CEA 1.59 ng/mL).\nTreatment was well tolerated, except for nausea (G2) and neutropenia (G2). Since the second cycle, granulocyte colony-stimulating factor (G-CSF) for secondary prophylaxis was administered on days 8-13 of each cycle.\nIn light of the young age of the patient and the stabilization of the disease we decided to reconsider the surgical approach, after a pre-operative abdomen MRI was performed (). On 06 April 2010 umbilical resection with the bladder dome and the urachal remnant was performed (). No postoperative complications arose.\nThe final histology report confirmed the diagnosis of urachal adenocarcinoma with mucinous components (CDX-2 positive, CK 20 positive, CK 7 negative, CK 34 beta E12 negative, beta-catenin positive); surgery was radical, with negative surgical margins. Follow up was then activated ().\nThe CT scan performed after two months showed the presence of two right lung nodules.\nOn 17 June 2010 the patient underwent precision resection of the 3 right lung nodules (for 2 of them, the CT scan was positive for metastasis from urachal carcinoma; the third, supposed by the surgeon to be malignant, resulted benign fibrotic parenchymal tissue). Surgery was radical and then follow up was started. A subsequent CT scan (24 August 2010) was negative for metastatic disease, although some small lesions, equivocal but suggestive of peritoneal involvement, were detected. Given both the absence of symptoms and the negativity of serum markers we decided, in agreement with the patient, to proceed only with clinical and radiological follow up. A CT scan performed after three months showed peritoneal disease in close contact with the uterus and bladder wall. Serum markers were elevated (CEA: 174 ng/mL, CA 19.9: 321 U/mL).\nThe patient was symptomatic for abdominal pain, therefore transdermal therapy with fentanyl (50 mcg/h) was started. Since the disease was progressing, we considered starting a second-line chemotherapy. Among the few chemotherapeutic regimens tested in this rare tumor, we chose a platinum/taxane combination. On 01 December 2010, we started chemotherapy with cisplatin (75 mg/m2: 130 mg tot.) and docetaxel 75 mg/m2 (130 mg tot.) day 1, q 21 for three cycles (until 18 January 2011). Primary prophylaxis with a single subcutaneous injection of pegfilgrastim was also performed. Treatment was well tolerated, with only nausea (G2) as adverse event; however, a CT scan performed after the end of the third cycle showed evidence of abdominal disease progression. Within few weeks since the end of chemotherapy, the patient reported moderate asthenia. Physical examination revealed paleness and a palpable, hard central pelvic mass (maximum diameter 5 cm). Hematological tests showed acute anemia (Hb 7.3 g/dL); the patient received transfusion of 2 units of red blood cells (05 February 2011) with return to satisfactory Hb values (Hb 10.5 g/dL on 08 February 2011).\nThe disease was progressing despite chemotherapy and surgery on both primary and metastatic sites. Nevertheless, the patient was in relatively good clinical conditions: vital signs were within the normal range and organ functions, as assessed by blood laboratory tests, were good. Given the young age of the patient, who was also a mother of two kids, we decided to try another treatment and to initiate off label therapy with the multikinase inhibitor sunitinib. On 23 February 2011 we started therapy with sunitinib at the dose of 25 mg continuously, with close clinical and hematological monitoring; after two weeks since the start of treatment the patient reported feeling better, with improvement of pain, no adverse events and good hematological profile. After about 50 days, analgesic treatment was discontinued because of pain disappearance; we performed a CT scan that showed stable disease, with evidence of necrotic evolution of the abdominal mass, which was also smaller and softer upon physical examination. The patient received sunitinib until May 2011, when she developed metrorrhagia. For this reason, sunitinib therapy was discontinued for seven days, with resolution of bleeding but also a prompt recurrence of abdominal pain. Treatment with sunitinib was started again, but metrorrhagia persisted. A radiation oncology consult was obtained, and hemostatic radiotherapy (total dose: 15 Gy in 3 fraction) was administered, with control of bleeding.\nThe patient continued sunitinib until 10 July 2011, when symptoms of intestinal obstruction appeared; treatment was finally stopped and the patient started only supportive care at home. She died on 29 July 2011. |
A 39-year-old woman with a history of cesarean section for first childbirth was admitted to the University of Tsukuba Hospital for a second pregnancy. At 40 weeks of gestation for postdate pregnancy, a left lateral episiotomy followed by a forceps delivery was performed, and a baby weighing 3.93 kg was born in good condition. A fourth-degree perineal laceration was found on perineal examination immediately after delivery. Briefly, a vaginal examination showed a 7 cm laceration in the episiotomy. Rectal examination showed a 3 cm laceration in the lower rectum, but it did not advance to the anal ring. The external and internal sphincter injuries were minor.\nThe first operation for perineal laceration was performed under spinal anesthesia in the lithotomy position on the day of delivery. A multilayer closure was performed followed by independent sutures of the rectal wall, musculature of the perineal body with sphincter, vaginal epithelium, and skin. All the sutures were interrupted stitches using 4-0 monofilament absorbable sutures. Antibiotics (cefmetazole) were administered prior to the surgery and used until 5 days after surgery. Wound dehiscence was not observed, and the patient was discharged 11 days after surgery. The patient did not show any symptoms of fecal incontinence, SSI, or rectovaginal fistula on the outpatient examination 1 month after surgery.\nFive months after the first surgery, the patient noticed perineal swelling and pain, followed by an uncontrollable passage of gas or feces at the perineum. A small pinhole with a discharge was noted at the left perineum, at the 5-O'clock direction of the vagina, and 5 mm from the vaginal orifice. Magnetic resonance imaging revealed a small fistula tract from the lower rectum, but an external opening or perianal abscess was not revealed (). Colonoscopy also showed a small orifice at the anterior wall of the lower rectum (above the dentate line), and indigo carmine poured into the rectum was discharged from the perineal opening (). Vaginoscopy did not reveal a fistula tract in the vagina. Finally, a rectoperineal fistula was diagnosed. No evidence of Crohn's disease was observed. Anatomical schema demonstrating the position of fistula tract was shown in .\nSeven months after the first operation, a second operation, transperineal repair for rectoperineal fistula was performed (, ). Briefly, the patient received general anesthesia in the lithotomy position. A Lone Star Retractor System (CooperSurgical, Inc., Trumbull, USA) was attached to the anus and vagina. A 2 mm probe was advanced through the rectoperineal fistula to guide the route of the fistula. A 4 cm transverse incision was made on the perineal skin, 5 mm from the vaginal orifice. Dissection of the rectovaginal septum was performed widely, separating the vagina and rectum, and 2 cm of the entire fistulous tract was excised. A three-layer closure (rectum, perineal body, and vagina, respectively) was performed with interrupted sutures using 4-0 monofilament absorbable sutures. The subcutaneous tissue and skin were sutured horizontally; thus, the incision was changed in the vertical direction, keeping thick perineal tissue. A trans-anal drain was placed until 5 days after surgery. Antibiotics (flomoxef sodium) were administered prior to the surgery and used until 3 days after surgery. The patient was discharged 10 days after surgery. The patient did not show any symptoms of SSI, rectovaginal fistula, or fecal incontinence on the outpatient examination 6 months after surgery. Anorectal manometry showed a normal anal sphincter function. Colonoscopy showed no dip or fistula on the rectal mucosa. |
A 36-year-old male with no known comorbidities presented to the emergency room with the complaints of sudden onset aphasia, persistent vomiting, and bilateral lower limb weakness for the past 1 day. On examination, he had a Glasgow Coma Scale (GCS) of 11/15 (E3V2M6). His proximal and distal lower limb powers were 3/5. Rest of the examination was unremarkable. His history was significant for an episode of seizure 1 month back, with symptoms of altered mental state during the fit that resolved spontaneously within a few minutes. Computed tomography (CT) angiogram [] revealed a compact AVM in the left frontal lobe, three aneurysms in the DACA distribution, intraventricular hemorrhage involving the 3rd and 4th ventricles, venous drainage to large vein draining the superior sagittal sinus, and poor vascular blush in the territory of the left ACA, suggestive of an infarct. He was admitted to a high dependency unit for observation.\nThe patient was awaiting digital subtraction angiography for almost 2 days when his GCS suddenly dropped to 5/15, with pupils bilaterally equal, dilated, and nonreactive. He underwent emergency intubation and was shifted to the intensive care unit (ICU). Head CT without contrast and CT angiogram revealed redemonstration of subarachnoid hemorrhage from the proximal largest aneurysm with intraventricular extension predominantly along the corpus callosum, with effaced basal cisterns and an interval increase in both hydrocephalus and cerebral edema []. The patient was shifted to interventional radiology suite for emergency coiling of the aneurysm and subsequent insertion of an external ventricular drain (EVD) for secondary hydrocephalus in the space of the next 5 h following decreased consciousness. The decision to prioritize coiling over EVD placement was influenced by the risk of repeat hemorrhage from suddenly decompressing the ventricles.\nAngiography revealed a Spetzler-Martin Grade 2 AVM, being fed by two vessels, and draining into the superior sagittal sinus through the superficial cortical veins. The major feeder arose from left callosomarginal artery and a minor feeder from the left MCA. The nidus measured 18 × 14 mm in diameter. The angiogram also revealed multiple flow aneurysms on the feeder from left callosomarginal artery, with the largest aneurysm, 14 × 8 mm in size, at the callosomarginal bifurcation. Pericallosal artery, with a relatively sluggish blood flow, was seen arising from its neck, because of which this aneurysm was not embolized. The nidus and rest of the flow aneurysms were embolized using the onyx [ and ].\nThe immediate postprocedural period was uneventful and uncomplicated. The patient, nevertheless, did not regain consciousness and was shifted to the ICU on controlled mechanical ventilation for further management and observation. Postoperative angiogram did not reveal any significant spasm. However, the patient’s neurological status did not improve, and he died in the hospital following a complicated clinical course. |
A 15-year-old male patient presented to the trauma emergency room intubated in the field with multiple gunshot wounds. On the primary survey, the patient was tachycardic and intubated. The patient's heart rate ranged from 100 to 120 bpm, sinus tachycardia, and blood pressure 100/60 mm Hg. In the secondary survey, the patient had multiple gunshot wounds to the chest, abdomen, bilateral lower limbs, and left upper extremity. The patient had palpable pulses in the bilateral lower extremity and left upper extremity. In the trauma emergency room, the patient had left tube thoracostomy for a left hemothorax performed bedside. The patient underwent computed tomography imaging suggestive of pneumoperitoneum, an X-ray and angiogram of left upper extremity suggestive of left upper extremity comminuted humerus fracture and left upper limb ulnar artery injury (Figures , ). The patient underwent damage control exploratory laparotomy. Diaphragmatic injury and bladder injuries were repaired primarily, small and large bowel left in discontinuity, and for pelvic wall bleeding, preperitoneal packing was performed. Abdomen fascia was left open with temporary closure performed using the wound VAC (vacuum-assisted device). For left humerus fracture with an expanding hematoma fasciotomy of the left upper limb was performed. On completion of the left upper limb's fasciotomy, there were no radial or ulnar pulses for Doppler signals.\nAn exploration of the brachial and ulnar artery revealed a disruption in the ulnar artery with a 5-cm arterial segment loss secondary to the gunshot and bullet fragments. An effort to mobilize the blood vessel did not permit a tension-free anastomosis. The artery was 2 mm in diameter. A Fogarty embolectomy showed good flow proximally, however weak backflow. At the time the patient had worsening acidosis and exsanguination, the ulnar artery was shunted with an argyle 8 Fr shunt (Figure ). The patient had Doppler signals in the left upper extremity after the shunt. The patient had forearm and hand fasciotomy completed of his left upper limb. The patient underwent resuscitation followed by, angioembolization of the left internal iliac artery for pelvic bleed. Once hemoglobin was stable and base deficit corrected, the patient's left upper limb was re-explored, and the shunt removed and basilic vein interposition graft performed (Figure ). The patient continued to have good pulses distally. The patient had an external fixation of his left humerus fracture, and a relook laparotomy to remove the pre-peritoneal packs placed earlier and perform bowel anastomosis. The patient received no anticoagulation, given his high risk of bleed in the pelvis and abdomen. The patient underwent surgeries for his wounds and went on to have complete vascular recovery from the interposition graft. |
A 2-year-old healthy boy underwent an office-based surgical circumcision by his physician. During the procedure, profound bleeding was observed that was not controlled by applying direct pressure. The physician attempted to control the bleeding by multiple deep suturing; the bleeding was stopped successfully and the patient was discharged home. During the postoperative period, the child had progressive painful and difficult voiding with only few drops of urine after straining. During this period the child was prescribed painkillers for his discomfort and no additional evaluation was done. After about a week he was referred to a district hospital with fever (temperature=38.1C), agitation, vomiting, urinary retention, and significant abdominal distension. Abdominal examination showed decreased bowel sounds, dull percussion, and severe guarding. Urgent abdominopelvic ultrasound revealed large volume of free fluid in the abdomen and pelvis, with small amount of urine in the bladder. The initial lab tests also showed leukocytosis with left shift and increased blood creatinine and blood urea nitrogen. Catheterization with a 6-Fr feeding tube failed because it did not pass beyond the subcoronal urethra. The patient underwent emergency midline laparotomy with the diagnosis of acute abdomen and the fluid was drained. A small intraperitoneal bladder rupture was noticed at the dome of bladder. The gastrointestinal tract was inspected precisely and was intact. The diagnosis of intraperitoneal bladder rupture with urinary ascites was made probably due to near-total urethral obstruction. The rupture site was repaired in two layers and a suprapubic cystostomy catheter was fixed.\nTwo months later, the patient was referred to our center for further evaluation and treatment. Antegrade voiding cystourethrography (VCUG) was performed via the suprapubic catheter that showed terminal urethral obstruction (). Urethroscopy was attempted under general anesthesia that failed due to complete obstruction at 1 cm from the meatus.\nDecision was made to explore the area and to repair the urethra. Through a circumferential incision, distal urethra was elevated from the corpus spongiosum. A 3-Fr ureteric catheter also did not pass the obstructed part (). A 5-mm fibrotic tissue was encountered at the site of obstruction (). The corpus spongiosum was dissected free from the corpora cavernosa to prevent iatrogenic chordee after end-to-end urethral anastomosis. The obstructed fibrotic part of urethra was completely resected and an end-to-end urethral anastomosis was performed along with spongioplasty over an 8-Fr silicon catheter in two layers; dartos pedicled flap was used to cover the site of anastomosis. The postoperative period was uneventful and the patient was discharged home with suprapubic and urethral catheter. The urethral catheter was removed seven days following the surgery. The suprapubic catheter was removed four weeks after the surgery following normal urethral voiding and normal ultrasound. A VCUG was performed six weeks after the surgery that showed normal bladder and urethra with acceptable voiding per urethra (). During a 2.5-year follow-up period, the patient was asymptomatic with normal renal function, ultrasound, and voiding pattern. He had a maximum flow rate of 15.3 ml/sec in uroflowmetry study. |
A 65-year-old man with severe chronic obstructive pulmonary disease (COPD) was admitted with a four-day history of chest pain and worsening shortness of breath. He explained the chest pain started suddenly when he tried to reach out for something on his computer table. It was located on the right anterior chest, sharp in nature, 7/10 in intensity, pleuritic, and worse with coughing and deep breathing. He had a past medical history of severe COPD with frequent exacerbations recently necessitating multiple antibiotics and steroid courses, coronary artery disease, gastroesophageal reflux disease (GERD), and hypertension. He had a 30-pack per year smoking history and quit about 10 years ago. He was a retired fireman living with his family.\nVitals signs in the emergency department (ED) were stable; he was breathing on ambient air. The physical examination demonstrated decreased breath sounds bilaterally without any wheezing or Ronchi. Moderate tenderness was present in the mid-axillary line in the fifth intercostal space, but no other abnormalities were noticed. Laboratory investigations were negative for any leukocytosis, troponin, or any other abnormalities. The electrocardiogram (EKG) showed a normal sinus rhythm. Computed tomography (CT) angiography was done to rule out pulmonary embolism (PE). The CT was negative for PE but showed mild peribronchial infiltrate in the right middle lobe and posterolateral lung herniation between the seventh and eighth ribs, with minimal subcutaneous emphysema along the right chest wall (Figure ).\nThe patient was admitted to the hospital and managed conservatively on broad-spectrum antibiotics, including vancomycin, levofloxacin, and 40 mg per day of prednisone. Two days later, his face swelled up suddenly with a change in the quality of his voice while he was eating dinner. An examination showed a swelling in the neck, diffuse crepitations on his body involving the face, all the way down to the buttocks. Repeat CT chest and neck showed extensive subcutaneous emphysema in the face, neck, chest, and mediastinum with a right-sided pneumothorax at the level of the previous lung herniation (Figures -).\nThe prevertebral and retropharyngeal air was demonstrated as compressing the oropharynx (Figure ). His oxygen requirement went up to 6 liters nasal cannula. A blowhole incision was made on the anterior chest wall, and he was observed in the medical intensive care unit (MICU). Surgery to close the defect was deferred due to his other comorbidities and the higher risk of post-operative complications. He improved gradually over the course of the next few days, completed the course of antibiotics with steroids, and was discharged to a rehabilitation center and did well post-discharge. |
The patient is a 4-year-old boy, weighing 17.5 kg, with no relevant medical or surgical history and no documented allergy or medication intake, scheduled for circumcision. His preoperative vital signs were a heart rate 98 beats/min, blood pressure 100/56 mmHg, temperature 36.6°C, and a respiratory rate of 20/min. Chest was clear to auscultation and no cardiac murmur was identified. The anesthetic plan was explained to the parents and included general anesthesia and a penile block for postoperative pain management.\nAnesthesia was induced in the presence of the parents with oxygen/nitrous oxide and sevoflurane administered through a face mask, with the patient breathing spontaneously. Propofol 40 mg and fentanyl 25 μg were administered after intravenous access. Anesthesia was maintained with sevoflurane at an end tidal concentration of 3.5% in a 1 L equal mixture of oxygen and air after securing the airway. The heart rate was noticed to be in the range of 80 beats/min, dropping to 60 beats/min. Ringer's lactate solution was administered at 10 mL/kg/h. A close observation of the electrocardiogram (ECG) tracing revealed a complete dissociation of P wave and QRS complex []. Blood pressure was normal and the patient was normothermic. The surgeon was notified, and resuscitation equipment was brought to the room. A trial of switching from sevoflurane to desflurane did not produce any change in the heart rate and was equally tolerated by the patient. Inhalation agents were discontinued and propofol infusion started at 300 μg/kg/min for the remaining 25 min of the procedure. There was no change in hemodynamics or in the ECG tracing. Atropine 250 μg was administered intravenously for a heart rate below 60 beats/min with a transient effect of increasing the heart rate to 80 beats/min. A penile block, with 10 mL of 0.25% bupivacaine was performed at the end of surgery. The patient was transferred awake to the recovery room. An urgent 12 lead electrocardiogram with cardiology follow up were requested.\nIn the recovery room the heart rate averaged 58 beats/min and the electrocardiogram tracing showed a third-degree AV block with a QTc of 450 ms. Patient was admitted for further evaluation. The structure and the function of the heart were within normal range by echocardiography. Laboratory studies to rule out systemic inflammatory and connective tissue diseases were negative. A Holter monitor showed a third-degree heart block with maximum heart rate of 95 beats/min and slowest heart rate of 35 beats/min with an average heart rate of 52 beats/min. The heart rate was below 70 beats/min in 97% of the times during the Holter monitoring. The patient had a dual chamber pace maker placed after a third-degree AV block and episodes of bradycardia as low as 30 beats/min were read on a repeat postoperative ECG and Holter monitor, respectively. |
The patient was a 56-year-old female with a history of type two diabetes mellitus, atrial fibrillation, mild aortic stenosis, breast cancer treated by resection and radiotherapy, and end-stage renal disease. She had undergone two failed renal transplants. For the last six years, she had received hemodialysis as renal replacement therapy. She had been receiving hemodialysis three times per week through a femoral line that had been inserted one year prior to her current presentation. Of note, her past medical history also included gram-positive rod bacteremia related to tunneled CVC infection two years prior. The causative organism turned out to be a Bacillus species.\nThe patient presented to the emergency department with subjective fever, chills, and fatigue with no other focal symptoms. She was hypotensive with a blood pressure of 81/39 mm Hg, but other vital signs were within normal limits (temperature: 36.9° C, pulse rate: 95 beats/minute, oxygen saturation: 97% on room air). Cardiac examination showed normal first and second heart sounds, and an ejection systolic murmur grade of 3/6 was heard most loudly in the aortic area and radiated to the carotids. There was no evidence of an exit site or tunnel infection at the site of CVC, and there were no peripheral stigmata of infective endocarditis.\nLaboratory studies showed a white cell count of 5,360 per microliter, hemoglobin level of 12 g/dL, and platelet count of 199,000 per microliter. Other basic workups, including liver and renal function tests, were within her baseline. Blood cultures were taken (central and peripheral samples), and the patient was started empirically on vancomycin and amikacin. Later, the aerobic bottles of both central and peripheral blood cultures taken initially turned out to be positive for gram-positive rods. Amoxicillin was added to cover the possibility of Listeria infection. A good-quality transthoracic echocardiogram was performed and showed severe aortic stenosis and no evidence of vegetation (Figure ).\nAll seven blood cultures taken over a 10-day period were positive for gram-positive rods. The bacteria grew as yellow-pigmented colonies within 48 hours of incubation at 35-37 °C under 5% CO2. The isolate was oxidase- and catalase-positive. The isolate was loaded on a Vitek2 ANC card (BioMérieux SA, Marcy-l'Étoile, France) according to the manufacturer’s protocol, but Vitek2 failed to provide any identification (software version 7.01). The isolate was then tested in triplicate with Vitek MS matrix-assisted laser desorption/ionization time-of-flight Mass Spectrometry (MALDI-TOF) (BioMérieux) as per the manufacturer’s protocol, but the Vitek MS also could not identify the organism, and hence the sample was sent to a reference laboratory for identification (Mayo Clinic Laboratories, Rochester, MN); the organism was identified by 16S polymerase chain reaction (PCR) as belonging to Leifsonia species. Table shows the susceptibility level of the organism using E-tests. It was interpreted according to the Clinical and Laboratory Standards Institute (CLSI) guidelines, which have it under related coryneform genera. Based on the final identification and biogram, amoxicillin and amikacin were stopped.\nThere was a delay in removing the line despite the persistent bacteremia, as the patient had very difficult vascular access with superior vena cava stenosis due to multiple catheter insertions. In addition, previous trials of angioplasty had failed. Eventually, the femoral line was removed, and a new right femoral line was inserted three days later. A negative blood culture at 72 hours of incubation was confirmed before inserting the new line. All blood cultures repeated after removing the line were negative. The patient improved clinically as her symptoms resolved and her blood pressure stabilized. She was discharged with the advice to continue on vancomycin intravenously for four weeks from the first negative blood culture and to maintain a vancomycin trough level between 15-20. The possibility of infectious endocarditis could not be excluded by transthoracic echocardiogram. However, we did not opt for a transesophageal echocardiogram as it is an invasive procedure, and we decided to treat her with a long course of vancomycin. The patient was seen in the infectious diseases clinic three weeks after completing the antibiotic course. She reported feeling well and had no symptoms suggestive of infection. Two sets of peripheral blood cultures were taken at that time, which did not grow any bacteria. The hemodialysis center, where the patient received dialysis sessions, had no other cases of Leifsonia infection or gram-positive rod bacteremia. Raising awareness among the hemodialysis unit personnel was conducted, and they were informed that this organism could be transmitted by contaminated dialysate water. We advised the personnel to report any further cases. |
A 17-year-old boy with history of fall from height of approximately 15 meters presented to our institution after receiving primary care at another hospital. At presentation, he was conscious, hemodynamically stable maintaining oxygen saturation at 98% on room air with no visible signs of respiratory distress. His Glasgow coma scale (GCS) was 15, and was able to move all four limbs. The patient had sustained open fracture both bone left leg along with fracture right ankle.\nChest radiograph showed no intrathoracic injury with normal lung parenchyma. Computed Tomography showed burst fracture of fifth lumbar vertebra with canal compromise () and ruled out any injury to head, cervical spine, thorax and abdomen. After primary care, patient was admitted to the orthopedic ward for spine stabilization surgery and surgery for lower limb fracture.\nOn day four post-admission, an emergency consultation call was sent to our intensive care unit (ICU) in view of patient’s deteriorating status. When seen, he was grossly pale and febrile at 101 F, pulse rate of 140 per minute, systolic blood pressure of 80 mmHg and respiratory rate of 32 per minute maintaining oxygen saturation around 90% on oxygen face-mask. Patient’s GCS was 15. Chest auscultation revealed bilateral diffuse coarse crepitation and he was immediately transferred to the ICU. Initial arterial blood gas (ABG) showed partial pressure oxygen (pO2) of 49mm Hg on oxygen by face mask. Patient was intubated, sedated, paralyzed and put on mechanical ventilation with initial settings of volume assist control and high positive end expiratory pressure (PEEP). Central venous catheter was secured in right internal jugular vein under ultrasound guidance. An arterial line was secured in right radial artery for invasive blood pressure and arterial blood gas analysis. Chest radiograph showed bilateral fluffy opacities (). Preliminary blood investigations were mostly unremarkable except for hemoglobin of 6.8 mg/dl and raised ESR of 44. Fundus exam specific for FES was normal and there was no petechial rash on general examination. Urine for fat globules was positive. Over the next few hours, patient’s hypoxemia worsened requiring higher fraction inhaled oxygen (FiO2) of up to 0.8 and PEEP of 16 cm water. A decision was made to turn the patient into prone position after discussion with the orthopedic surgery team in view of unstable lumbar spine fracture. Patient’s family were informed about the specific risks and benefits of prone positioning, particularly in a patient with pre-existing unstable lumbar spine fracture and a written consent for the same was obtained. Positioning was done with the help of 5 trained ICU staff, using logrolling technique for turning the patient lateral followed by a 6th member placing the spinal board under the patient. After securing the patient on the spinal board, he was then shifted to one edge of the bed while the head gel support, chest and pelvic roll were placed in position. Patient was then shifted to prone position and spinal board was removed. Patient’s hands were abducted and placed next to the head and all the pressure points were padded using pillows and cotton rolls.\nOver the next 16 hours, we kept our patient sedated and paralyzed in prone position during which he received targeted fluid therapy and remained hemodynamically stable. Wake up test and pupil examination were done at regular intervals. Serial ABG’s showed dramatic improvement with Fio2 requirement decreasing to 0.4 and PEEP of 8 following which patient was repositioned supine. Two units packed red cells were transfused while the patient was prone. Chest radiograph showed resolving lung infiltrates and we decided to electively mechanically ventilate our patient for next 24 hours in supine position. Weaning was started next day and patient was extubated a day after and transferred back to ward 24 hours later.\nThe patient later on went on to have multiple surgical procedures for injuries to spine and bilateral lower limbs. He was then followed for 8 weeks in the out-patient department following discharge during which he recovered well without any neurovascular deficit. |
A 13 year old boy presented with complaints of intermittent fast palpitations of 3 months duration. The sinus rhythm ECG showed notching of T waves in V2-4 but was otherwise normal (). The ECG during tachycardia suggested a ventricular tachycardia (VT) with left bundle branch block (LBBB) morphology () which was responsive to intravenous diltiazem and lignocaine when given at different occasions elsewhere. Even on amiodarone and diltiazem at maximum tolerated doses a 24 hour Holter showed presence of several runs of non-sustained VT (NSVT), though at slower rates and multiple premature ventricular complexes (PVCs). The echocardiogram was normal. Hence radiofrequency ablation was planned. After sedation with midazolam 1 mg and catheter placement there were no ectopics, NSVT or VT either spontaneously or on pacing protocols, even after isoprenaline. After four hours of observation, NSVT and PVCs reappeared.\nConsidering non-inducibility of arrhythmia due to sedation, a restudy without sedation was planned. There were frequent PVCs and NSVT just prior to the study. However again after preparation and giving local anaesthesia, not even a single PVC was seen. That is when we realized that the patient's arrhythmia could have been extremely sensitive to lignocaine which was given subcutaneously as a local anesthetic agent. After 4 hours again the PVCs reappeared. To confirm our hypothesis, subcutaneous lignocaine (local anaesthetic) was administered in cardiac care unit (CCU). This suppressed the PVCs within 2 minutes () and the effect lasted for 4 hours. Subcutaneous bupivacaine also suppressed the PVCs for nearly 6 hours.\nRestudy was undertaken without giving local anesthetic; only sedation with 2 mg midazolam intravenously was used. A single femoral venous puncture was taken considering VT morphology, and an ablation catheter was used to map during NSVT and PVCs, which this time, did not get suppressed. The site of earliest activation was at a site near the His bundle (). Pace-map showed a 12/12 match at the same site (Figure 3b). Continued pacing caused intermittent His bundle capture with a narrowed QRS (). Ablation was hence not considered safe. A trial of intravenous phenytoin and metoprolol did not result in any decrease in the ectopics.\nThe patient still continues to have NSVT and VT even after giving diltiazem, though at a lesser frequency and rate, which respond immediately to lignocaine. The patient and relatives have been instructed to administer subcutaneous lignocaine in case of emergency and to consider ablation if significant symptoms persist, but with high chances of developing complete atrioventricular block during procedure. |
A 60-year-old male with metastatic RCC treated with nivolumab and palliative radiation therapy presented to our institution in 2016 with shortness of breath and was found to be in acute respiratory failure. Computed tomography (CT) of the chest was significant for multiple new ground-glass opacities throughout bilateral lungs concerning for therapy-induced pneumonitis (Figures , ). The etiology of ground glass opacities includes but is not limited to infectious pneumonitis, bronchioloalveolar carcinoma, or interstitial disease. Given the timing of symptom onset as well as lack of response to infectious treatment, therapy-induced pneumonitis remained high on our differential.\nHe initially presented in 2011 with gross hematuria and right-sided flank pain and underwent right radical nephrectomy and lymph node dissection of a 9 cm Fuhrman grade IV RCC with negative margins and lymph nodes. Two years later, surveillance imaging and biopsy were significant for metastatic RCC in the lungs. He was initially treated with one year of sunitinib, a multi-targeted receptor tyrosine kinase inhibitor. However, given the progression of disease, he was transitioned to one year of pazopanib followed by six months of axitinib, one month of everolimus, and five months of sorafenib. Pazopanib, axitinib, and sorafenib are also tyrosine kinase inhibitors. Everolimus is an inhibitor of mammalian target of rapamycin. Given the lack of response to these therapies, our patient was started on nivolumab at 3 mg/kg in May of 2016. Over the course of four years, he received targeted palliative radiotherapy including 1900 centigray (cGy) to a left upper lobe lung mass in May 2016 and 800 cGy to an L5 lesion in September 2016.\nHe complained of chronic shortness of breath for three months felt secondary to anemia and a left pleural effusion before presenting to our hospital in acute respiratory failure with CT evidence of new diffuse ground-glass opacities occupying the majority of both lungs (Figures , ). Given high suspicion for therapy-induced pneumonitis, he was started on a treatment course of high dose steroids. However, the patient’s respiratory status continued to decline and he passed away on comfort measures.\nPathology was significant for organizing diffuse alveolar damage with hyaline membrane formation in all lobes of both lungs away from the metastatic RCC (Figures , ). There was no evidence of an infectious process from cultures and pathologic evaluation. This histologic reaction pattern is a typical finding in patients with a clinical diagnosis of acute respiratory distress syndrome (ARDS) concerning for therapy-induced pneumonitis. |
A 10-day-old male infant was referred to our hospital because of suspected congenital hypothyroidism. The patient presented with symptoms of airway obstruction, such as an inspiratory stridor and retracted breathing. A hormonal test revealed subclinical hypothyroidism with a free thyroxine level (1.44 ng/dL) within the reference range, although the thyroid stimulating hormone (TSH) level (34.6 μIU/mL) was increased beyond the normal range. Laryngo fiberscopy revealed a lingual mass compressing the epiglottis (Fig. ). Enhanced computed tomography (CT) and thyroid scintigraphy revealed that the mass was an ectopic thyroid with the absence of a normal pretracheal thyroid gland (Figs , ). The patient received oral levothyroxine at a dose of 12 μg/kg/day for 4 weeks to lower the TSH level and reduce the volume of the ectopic thyroid tissue. However, we observed no reduction in the volume of the thyroid tissue and a concomitant progression in his symptoms of airway obstruction. He underwent surgery to relieve the airway obstruction when he was 2 months old. Under general anesthesia, nasotracheal intubation was performed in a sniffing position, and a transverse skin incision measuring 2.5 cm was made at the level of the hyoid bone. We split the hyoid bone at the midline, dissected the base of the tongue towards the foramen cecum, detected the ectopic thyroid mass, and suspended the mass by suturing it to the hyoid bone (Fig. ). We used 5–0 monofilament absorbable sutures and sutured between the lingual thyroid and the hyoid bone. The bite length of both the lingual thyroid and hyoid bone was about 3 mm. The points of suturing were to the lateral side of the lingual thyroid and to the front of it. The total number of suture threads was 3. The degree of suspension of the ectopic thyroid was guided by an intraoperative laryngo fiberscopy to confirm the complete elevation of the epiglottis. The patient was not extubated until postoperative day 4 and needed noninvasive positive pressure ventilation until postoperative day 22. Laryngo fiberscopy performed 6 months postoperatively revealed the complete disappearance of compression of the epiglottis by the lingual mass, and CT performed 8 months postoperatively also revealed the relocation of the lingual thyroid gland towards the hyoid bone (Fig. ). When the patient was 2 years 6 months old, his height was 94.1 cm(1.4 SD), weight was 14.0 kg(1.0 SD), free T3 was 2.97 pg/mL, free T4 was 1.48 ng/dL, and TSH was 4.178 μIU/mL. He was taking daily levothyroxine 4.5 μg/kg/day and had been kept in the euthyroid state. Since we were able to preserve his thyroid gland (which is his only functioning thyroid tissue), the postoperative control of his thyroid hormone status was relatively easy. The surgery was complicated by the development of a salivary fistula that was spontaneously resolved 5 months postoperatively. |
A 53-year-old male was referred to our hospital in September 2012 for re-transplantation due to severe biliary complication after the first LTx. His blood type was “O”. Complete SI without other abnormalities was identified when he was a teenager. Following splenectomy and hepatectomy due to hemorrhage after hepatitis B related cirrhosis and hepatocellular carcinoma respectively, he underwent LTx from a deceased donor with situs solitus because of tumor recurrence in 2005 at the Eastern Hepatobiliary Surgery Hospital, Shanghai, China. Since half a year after LTx, he suffered episodes of biliary complication characterized as jaundice and fever. Bile duct stents were inserted several times. The total bilirubin fluctuated between 70-200 μmol/L and it was gradually elevated to about 400 μmol/L before he was listed for re-transplantation. He had severe skin pruritus and sporadic slight hepatic comma.\nThe donor was a 32-year-old male who suffered brain injury in a motor vehicle accident and was pronounced dead shortly after admission. Image studies showed that he was situs reversus. His blood type was “B”. Given the organic situs match, the donor liver graft was then allocated to the recipient. No technical difficulties were encountered during the procurement. The liver graft showed completely inverted structure but without vessel anomaly after trimming the excess tissue on back table (Figure ).\nAfter careful laparotomy in recipient, a normal oriented liver was transplanted showing deeply bilious appearance with blunt right lobe and shrunken left lobe. The whole graft was severely rotated to the left upper quadrant. Excision of the original liver graft was performed with preservation of recipient inferior vena cava (IVC). The new liver was then brought onto the field as its original position. The structure of IVC, hepatic artery, portal vein and common bile duct of the graft were in line with the recipient’s corresponding structures. A piggy-back technique with some modifications was applied for cavo-cavostomy. In brief, a longitudinal midline incision on the posterior wall of donor suprahepatic IVC was cut to make a triangulated and wide orifice, which was end-to-side anastomosed to the corresponding similar incision in the anterior wall of recipient IVC from the orifice of hepatic veins. Donor infrahepatic IVC was ligated. The portal vein was end to end anastomosed. The donor’s common hepatic artery was anastomosed to the confluence of the recipient’s proper hepatic artery and gastroduodenal artery. Cholecystectomy was performed as usual and biliary reconstruction was achieved via end-to-end choledochocholedochostomy (Figure A, B).\nTo reduce the risk of complications from ABO mismatch, the patient was treated with the protocol of our center: 600 mg Rituximab (MabThera®, Roche) before transplantation and a total of 200 g intravenous immune globulin during operation and the first ten consecutive days post transplant. The maintenance immunosuppression included tacrolimus, mycophenolate mofetil and steroid. The post-operative outcome was uneventful. He was doing well with excellent graft function at 11 months follow-up. Computed tomography assessment demonstrated the graft in normal appearance in the up left quadrant (Figure ). |
An 83-year-old-woman presented with a subcarinal mass detected by a precise examination of upper abdominal pain worsening after meals. Magnetic resonance imaging revealed a well-circumscribed cystic mass in the subcarinal area compressing the surrounding organs and suggested a BC as the most likely diagnosis (). She had poorly controlled type 2 diabetes mellitus (A1c level 9.1%), hypertension, and also noted worsening paroxysmal atrial fibrillation (CHADS2 score3) and was receiving dabigatran etexilate over the last several months. A surgical resection of the cyst was recommended. With the patient in the prone position, two 5-mm ports in the third and fifth inter costal spaces (ICSs) and a 12-mm port in in the seventh ICS on the mid-axillary line were made, respectively. An additional 5-mm port was inserted into the eighth ICS on the scapular line and used as a camera port (). Carbon dioxide (CO2) was insufflated at a pressure of 8–mm Hg and then a tense mass was clearly visualized in the subcarinal area (). The cyst wall was found to have strictly adhered to the adjacent organs suggestive of past inflammatory events. Because an attempt to dissect between the cyst and lung resulted in parenchymal injury with alveolar leakage, we considered that the lesion was not amenable to a complete excision. After\nthe fenestration of the cyst wall with the aspiration of an yellowish mucus content, a large proportion of the thickened wall was removed to open the cavity wide enough. The remnant luminal epithelium was ablated by electrocautery. The operative time was 117 minutes and the bleeding amount was 5 g. The postoperative course was uneventful, and she was discharged on the second postoperative day. A histopathological examination revealed a ciliated epithelium, hyaline cartilage and bronchial mucus glands consistent with a BC. Her digestive problem and palpitations disappeared immediately after the surgery, and she is currently symptom free at 18 months. |
A 16-year-old male presented to medical attention with a history of intermittent headache since last 1 month, particularly after doing strenuous activity. Patient advised for computed tomography (CT) scan [] of the brain that showed a large mixed density lesion with mass effect for which he was referred to us. On examination patient was conscious, cooperative, and oriented to time, place, and person. His vitals were within normal range. He had no significant medical history, previous radiation exposure, or family history of vascular malformations. There was no sensory or motor deficit. His both fundus appears to be normal. Magnetic resonance imaging (MRI) of the brain [] showed a large heterogenous partially hemorrhagic, multilobulated mass (68 mm × 69 mm × 74 mm) in right frontal lobe with perilesional edema and midline shift of approximately 15 mm to the contralateral side. Magnetic resonance spectroscopy performed and was nonconclusive for neoplastic pathology. CT angiography of the brain was negative for any Arteriovenous vascular malformation. After interdepartmental discussion and words with Neuroradiologist provisional diagnosis of arteriovenous malformation (AVM) with bleed or a high-grade malignancy with hemorrhage was made. After taking written and informed consent, the patient was taken for surgery. On operation table, his head end was elevated so that cerebral venous drainage improved. Question mark incision made and frontotemporoparietal craniotomy done. Dura opened after applying dural hitches. The brain was tense and edematous. A small opening was made over middle frontal gyrus, and cystic part of lesion aspirated out with help of suction. Cystic fluid was like old blood. Solid part of lesion was soft to firm in consistency and was purple green in appearance. Soft part of lesion was a suckable, but firm part was taken out in piecemeal. There was gliotic reaction all around the lesion that was left in situ. Overall, during surgery parts of the lesion bleed profusely with dark venous blood but controlled using bipolar cautery, hemostatic agents, and light pressure. Total excision of lesion was done. Immediately after surgery, the patient was arousable and moving all four limbs with obeying of commands. Postoperative period was uneventful, and the patient did not develop any neurological deficit. Histopathological examination [] confirmed the diagnosis of CM. The patient was discharged on 10th postoperative day. Postoperative follow-up to 6 years and there was no development of new neurologic symptoms recurrence of the lesion on imaging. |
The 35-year-old man underwent a renal transplantation four years before due to SCN-associated ESRD. His recent serum test was positive for donor-specific antibody, and his current serum creatinine was elevated to 3.8 mg/dL before the renal allograft biopsy.\nSections of light microscopy revealed two biopsy cores of renal cortex containing totally ten glomeruli (). One glomerulus was globally sclerosed and the remaining glomeruli showed membranoproliferative pattern with thrombi in one glomerulus. There was lymphocytic infiltration involving more than 20% of the renal cortex with mild tubulitis. No vasculitis was identified. Trichrome-stained section showed mild to moderate interstitial fibrosis and tubular atrophy. BK virus staining was negative with a good positive control. C4d staining by immunohistochemical method was strongly and diffusely positive in glomeruli and peritubular capillaries (). Iron staining was essentially negative in the renal tubules (). Three glomeruli were present for immunofluorescence studies. At least two glomeruli showed segmentally positive staining for fibrinogen (2+) and IgM (1+). The remaining staining including IgG, IgA, C3, C1q, kappa, and lambda was negative. C4d stained sections showed strong positive staining in 80% of the peritubular capillaries, consistent with antibody-mediated rejection. Four glomeruli were identified on Methylene Blue Azure II stained sections for electron microscopy. Ultrastructurally, there was segmental fusion of foot processes. The glomerular basement membrane showed prominent double contour with lucent appearance and scattered foci of subendothelial fibrin deposits (). Occasional irregular shaped RBCs were present, but they do not form thrombus. No immune complex deposits were identified in the mesangial areas, subendothelial or subepithelial spaces. We commented that the TMA can be either secondary to the antibody-mediated rejection or SCN. However, the negative iron stain in the renal tubules implied that SCN was not the major cause for this current TMA. Instead, the TMA was most likely associated with the antibody-mediated rejection and thus classified as type II acute antibody-mediated rejection. |
A 51-year-old female patient presented with severe radiating pain bilaterally in the posterior aspect if the thighs and legs of 3 months' duration. Six months prior to admission, she underwent an L4/L5 decompressive laminectomy with SML augumentation from L4 to L5 for chronic right leg radiating pain (L5 distribution) of 6 months' duration. The postoperative course was uneventful and the radiating pain in her right leg was relieved.\nHowever, left buttock pain radiating to the left thigh and leg developed gradually 3 months postoperatively and pain in the right thigh and leg subsequently reccurred. Her bilateral thigh and leg pain did not respond to restriction of activity and maximal medications including ultracet, tramadol, gabapentin (up to 1800 mg a day), and oxycodone (20-40 mg a day). Application of transdermal fentanyl caused dizziness and abdominal discomfort. Repeated root blocks (S1) and epidural blocks were effective only for several hours. A postoperative magnetic resonance imaging (MRI) taken at the time of pain recurrence showed a signal artifact along the SML and no evidence of recurrent disc herniation or lateral stenosis (). She was referred to the authors for further evaluation and management.\nOn examination, no motor weakness or sensory disturbance was detected. The deep tendon reflexes were intact and there was no urinary incontinence. Mild limitation in straight leg raising (60/60) was noted. There was no low back pain. Her pain, much more severe in the left side, occurred bilaterally in the lower buttock and radiated to the posterior thigh and legs along the S1 dermatomes. The radiating pain was aggravated with standing, walking and straining. Careful reading of the MRI taken at the time of recurrence suggested a possible dural compression from the SML in the opinion of the authors, and a computed tomography (CT) myelogram was performed. On CT myelography, the upper and lower distal ends of the SML bilaterally were found to encroaching on the posterior dural sac (), and removal of the SMLs was planned to relieve the bilateral leg pain after receiving informed consent.\nAfter re-opening of the previous operation site, the paraspinal muscles and fascia were dissected and retracted. The adhesions along the SML were carefully divided and the SMLs were exposed (). It seemed that the SMLs were firmly attached to the spinous processes and laminae, and no jolting movement of the SML was elicited by shaking with Kelly clamps. The SML could not be rotated or bent with several kinds of clamps, so the arms of the SML were drilled down with a diamond burr under microscopic vision (). The lower ends of the bilateral limbs of the SML could be pushed out after the drilling.\nHowever, the upper ends that were hooked in the upper L4 laminae could not be extracted manually and additional drilling of the upper medial part of the laminae was needed to create rooms for the movement of the hooked SML ends (). Upon removal of the ends of the SML, a small hole for the SML was noted (). We noticed that the ligamentum flavum which should be removed to prevent dural compression during application of the SML had been left intact. After securing hemostasis, the overlying wounds were closed in layers.\nThe postoperative course was uneventful. Her disabling, bilateral radiating pain was relieved 2 days after the operation and the medications could be withdrawn two weeks postoperatively. No symptoms related to possible instability of the laminectomized segment resulting from the removal of the SMLs were noticed until 6 months after the removal of the SMLs. |
A 37-year-old woman was admitted to our emergency department with the chief complaint of headache that has gradually been deteriorating in the last month and was accompanied by blurred vision. The headache was worst in the morning and was localized in the frontal and occipital region. The patient also mentioned two episodes of vomiting and phonophobia along with the headache the last week. Physical examination was normal, however the patient complained for mild unsteadiness and recent memory loss. No loss of bladder control or urgency to urination was mentioned. As far as the patient's past medical history is concerned, an Epstein-Barr virus (EBV) infectious mononucleosis was described. The rest medical history was unremarkable. For evaluation of her symptoms the initial workup included a brain magnetic resonance imaging (MRI) which revealed significant ventricular dilatation of lateral, third and fourth ventricles with subependymal edema, without any gadolinium enhancement of the ventricular system or obvious obstruction. A T2W TSE study was also performed and demonstrated a turbulent flow of cerebelospinal fluid (CSF) in the fourth ventricle and back flow of CSF through the Monro's foramen to the lateral ventricles (). The patient had also undergone fundoscopic examination that showed bilateral papilledema.\nBased on the above findings a decompression on the foramen magnum was decided having established an obstruction at that level. The patient underwent a suboccipital craniotomy with C1 laminectomy. An occlusion of Magendie's foramen by a thickened membrane was recognized and the membrane was incised and removed, thus restoring the normal flow of cerebrospinal fluid between the fourth ventricle and the cerebellomedullary cistern ().\nThe postoperative course was uneventful. The new fundoscopic examination in two weeks revealed a reduction in papilledema and the second MRI scan showed a marked decrease in the size of the ventricles, showing therefore that cerebrospinal fluid flow through the ventricular system and subarachnoid space was restored (). |
A Caucasian male infant was born by cesarean delivery to a 33-year-old mother at 34 weeks of gestational age. The pregnancy was complicated by the presence of insulin-dependent gestational diabetes and the prenatal diagnosis of bilateral ventriculomegaly. Further prenatal evaluation with fetal brain magnetic resonance imaging (MRI) study revealed the presence of right open-lip schizencephaly with bilateral ventriculomegaly. Furthermore, her last pregnancy was complicated by shoulder dystocia, and given this history, along with the macrocephaly noted in this pregnancy with concerns for cephalopelvic disproportion, a decision was made to proceed with cesarean delivery. The cesarean delivery was uncomplicated, and the infant needed routine care with no resuscitation. On postnatal physical examination, the infant was noted to have macrocephaly with a head circumference measuring 36 cm, which was at >99th percentile for the gestational age. Anterior fontanel was very wide open. In addition, significant sutural diastases were also noted on physical examination. Even though the infant appeared non-encephalopathic, the presence of generalized axial and appendicular hypotonia was noted. During the first postnatal week, rapid head growth with tense and bulging anterior fontanel along with worsening sutural diastases was noted. Distended scalp veins were visible. Postnatal MRI performed on postnatal day four revealed the presence of open-lip schizencephaly located in the right occipitoparietal region of the cerebral hemisphere, communicating with the right lateral ventricle and extra-axial CSF space. Posterior fossa CSF space, which was in continuity with extra-axial CSF space [], was markedly dilated causing a mass effect on the cerebellum and tonsillar herniation by approximately 1 cm []. Both lateral ventricles were asymmetrically dilated, with right lateral ventricular dilatation more marked than the left. Also, a pronounced right lateral ventricular dilatation caused a leftward midline shift. Syndromic evaluation, evaluation for intrauterine infection, and evaluation for prenatal teratogenic exposure evaluation through detailed history yielded negative results. The pediatric neurosurgical evaluation followed by right VP shunt placement was performed. The infant had an uneventful recovery following the surgery. Parental consent was obtained for reporting this case. |
A 27-year-old Caucasian woman was admitted to the Emergency Department of our Institution because of bloody diarrhoea – up to 10 bowel movements per day – during the last month, 3 weeks after quitting smoking. Physical examination showed no abnormalities but confirmed haematochezia on digital rectal examination. Colonoscopy showed continuous severe colonic inflammation with small ulcers from the anus to the descendent colon, classified as grade 3 in Mayo endoscopic sub-score and 3 points in Ulcerative Colitis Endoscopic Index of Severity (UCEIS); complete examination was not performed because of the risk of perforation. Empirical antibiotic treatment with ciprofloxacin and metronidazole, as well as oral and rectal mesalamine were started and partial symptomatic improvement was achieved. Venous thrombosis prophylaxis with subcutaneous enoxaparin, 40 mg per day, was started. At admittance, haemoglobin, white cell count, platelets, fibrinogen and C reactive protein (CRP) were within the reference range. Stool cultures were negative. Cytomegalovirus (CMV) infection was also ruled out in colonic biopsies (polymerase chain reaction – PCR – and, later, immunohistochemistry). As bloody diarrhoea persisted 48 h later, and histopathological examination of colonic biopsies showed crypt distortion, a mixed inflammatory infiltrate of the lamina propria and crypt abscesses suggesting the diagnosis of UC, intravenous methylprednisolone (1 mg per kg of weight, daily) was started. After 3 days of corticosteroids the patient achieved partial clinical response (6 bowel movements per day, Edinburgh index 2 points, CRP within the normal range); nevertheless, 2 weeks later infliximab therapy (5 mg/kg of weight) was started due to sustained clinical activity, with 10 bloody bowel movements per day and a progressive increase of CRP levels, up to 10 mg/dL. Three days after the first dose of infliximab, the patient presented a massive lower bleeding with haemodynamic instability and severe anaemia; CT scan showed active arterial haemorrhage from ascendant colon; a subsequent arteriography demonstrated active arterial bleeding from a colic branch of the superior mesenteric artery; selective transcatheter embolization with platinum microcoils (MicroNester©, Cook Medical) was performed with immediate technical success; nevertheless, the patient persisted with rectal bleeding 2 days after embolization, requiring laparoscopic subtotal colectomy and ileostomy. Pathological evaluation of the colon confirmed the diagnosis of UC. Eight days after surgery the patient was discharged. |
A 35-year-old man with a history of severe (score of 10 in the verbal numerical scale), shock like and throbbing pain in the right V1–V2 region, lasting for 5–10 s that increased on talking, chewing, smiling, with strong breeze and cold water while washing his face since last 6–7 years. He visited a dental clinic for the same, for which he has undergone extraction of the upper right posterior teeth (16). The pain did not subside even he visited various clinics in various places where he was medicated with tablet carbamazepine 200 mg eight hourly. He reported to Department of Oral and Maxillofacial Surgery, Modern Dental College and Research Centre, Indore. After taking a detailed history, the trigger zones were noted which were nasolabial fold, ala of nose, cheek region, supraorbital rim on right side of the face. Diagnostic block was given in the infraorbital and supraorbital regions on different occasions for which pain was relieved for several hours. This confirmed the infraorbital and supraorbital neuralgia. To rule out the possible etiology of the neuralgia, MRI of cranium was advised and there was no intracranial involvement of the nerve was found. The treatment of peripheral neurectomy was planned as he was on already medication since last 6 years. Blood investigations were within normal limits.\nThe patient was taken under GA with endotracheal intubation and the supraorbital nerve was exposed via upper eyebrow incision []. The nerve was identified and avulsed by twisting the nerve on the artery forcep []. Double layer closure was done with 3–0 vicryl and 3–0 ethilon. Then, the infraorbital nerve was exposed through intraoral approach by giving upper vestibular incision from 13 to 16 region []. The infraorbital nerve was avulsed in the same way []. The double layer closure is done. Postoperative recovery was uneventful. Patient has been relieved of pain since last 1 year. |
A 56-year-old male without any previous medical history presented to our emergency room (ER) with multiple traumas from a 10 meter fall in a construction field. Physical examination revealed a male patient with a body mass index in the normal range and an acutely ill looking appearance. His right lower leg and ankle were swollen and bruised, and he had a 2 cm laceration wound on the plantar aspect of his right foot. The patient's right ankle had limited range of motion due to pain. The patient had tenderness at the right anterolateral aspect of the mid lower leg and anterior aspect of the ankle. There was grade 1 anterolateral instability of the left ankle. The neurologic examination was normal. Based on the patient's clinical history and physical examination, the orthopedic surgeon suspected a fracture of the right fibular diaphysis and ligament injury of the right anterolateral ankle.\nInitial radiographs of the ankle in the anteroposterior and lateral views showed fractures at the diaphysis at the fibula and anterior lip of the tibial plafond (Fig. ). The patient was not able to undergo ankle Mortise view because of his limited range of motion due to extreme pain. In a subsequent lower extremity computed tomography (CT), the orthopedic surgeon in the ER noticed a segmental fracture of the right fibular shaft and the anterior lip of the tibial plafond.\nTo evaluate the ankle ligaments, a turbo spin-echo (TSE) two-point mDixon technique applied to an ankle MRI (Table ) was performed after procuring written informed consent. In addition to the fractures of the right fibular shaft and tibial plafond, this MRI demonstrated a tiny chip fracture of the lateral talar dome. A tiny wafer-shaped talar dome chip fracture fragment about 7 (anterior–posterior diameter) × 3 (head to toe diameter) mm was clearly delineated only in the sagittal T2-weighted mDixon opposed-phase MRI (Fig. B). In T2-weighted mDixon in-phase imaging, which is considered a conventional T2-weighted image, there was a definite focal wedge-shaped cartilage defect at the corresponding area. However, there was only focal and subtle cortical irregularity and the cortical step-off was not definite (Fig. C). In a T2-weighted mDixon water-only image, which is considered a conventional fat-suppressed T2-weighted imaging, the cartilage lesion and focal cortical irregularity were once again noted, and the subcortical bone marrow edema was additionally confirmed. In these 2 sequences, a fracture was suspected, but the radiologists could not fully delineate the fracture line (Fig. D). In T2-weighted mDixon fat-only imaging, there were dark signal alterations at the subcortical region, but these were not considered fractures (Fig. E). T1-weighted imaging was obtained in the axial plane, and the fracture line was not depicted in this plane (Fig. F). In a CT image reviewed by an experienced musculoskeletal radiologist, there was a lateral talar shoulder cortical fracture at the identical area where the chip fracture was noted (Fig. A) from the T2-weighted mDixon opposed-phase image. In addition, there was a grade 2 injury to the anterior talofibular ligament with severe subcutaneous swelling of the ankle.\nDuring ankle arthroscopy, there was a free floating osteochondral fragment about 4 x 8 mm at the posterolateral talar shoulder, which was removed with basket forceps (Fig. ), and microfractures were performed at the posterolateral talar cortical fracture site.\nThe patient did well after the arthroscopy with recovery of full range of motion after 2 months. |
In November 2018, a 30-year-old woman presented to the Department of Oral and Maxillofacial Surgery of Chonbuk National University Hospital with a complaint of pain and swelling in the left mandible that had appeared 10 days earlier. Eleven years ago, she was diagnosed with CFD (Fig. a, b) and had received bone contouring in the left zygomaticomaxillary complex and left mandibular region under general anesthesia in the same department (Fig. a, b). Postoperative healing was uneventful and 18 months postoperatively, there was no specific problem with the lesion (Fig. c). The patient had subsequently been lost to follow-up until November 2018.\nA review of medical history prior to November 2018 confirmed that she had not received any dental treatment or suffered trauma to the painful left mandible area in recent months. Her pain intensity rating was 4 points on the numeric pain rating scale. Clinical examination revealed slight swelling in the left midface and left submandibular areas, along with bony expansion from the posterior of the left mandibular angle to the inferior aspect of the #34 tooth. The patient did not complain of hypoesthesia or pain when pressure was applied to the area. During an intraoral observation, the swelling was found from the distal aspect of #33 to the mesial vestibule area in relation to #36. During the endodontic examination, tooth mobility and percussion reactions were not observed in #34, 35, and 36. Moreover, the electric pulp test (EPT) showed normal response from #34, 35, and 36. No evidence of gum inflammation, such as periodontal pockets or gingival sulcus swelling and bleeding, was found during the periodontal examination. Furthermore, in the panoramic view, the dental origin with the possible infection source was not observed (Fig. d).\nA well-defined multilocular radiolucent lesion in the left posterior mandibular region was identified on the panoramic radiograph, and the location of the lesion overlapped with the existing FD. In addition, amorphous calcified foci were observed inside the lesion (Fig. d).\nCone-beam computed tomography (CBCT) showed an expansive bone lesion with a ground-glass appearance spanning the left frontotemporal bone, crista galli, orbital wall, ethmoid bone, sphenoid bone, zygoma, pterygoid plate, and maxilla regions (Fig. a). An ill-defined (partially well-defined) irregular osteolytic lesion was observed inside the left mandibular lesion, and cortical thinning, buccolingual expansion, and cortical destruction were also identified (Fig. a).\nA decision to perform a marsupialization procedure was made to first control edema and pain, and second to take a biopsy. The marsupialization procedure was performed after an intraoral incisional biopsy of the area surrounding the #34 and #35 teeth, followed by root canal treatments on these teeth. The biopsy results revealed some evidence of chronic inflammation and that the lesion may be a bony lesion with inflammatory reaction rather than FD. During a 3-week observational period, the size of the lesion was unchanged according to clinical and radiological findings (Fig. b). Eventually, we decided that cyst enucleation under general anesthesia should be performed in the mandibular region. However, in the maxilla region, since there was no pain or discomfort, we decided to follow-up without any surgical treatment.\nAt the time of surgery, the lesion had expanded from the inferior aspect of the #34 tooth to the mesial root of the #36 tooth, with fibrotic tissue scattered within the lesion. Subsequently, the soft tissue lesion was removed by cyst enucleation. The perilesional bone and the roots of the #34, 35 teeth, and the mesial root of the #36 tooth were ground. Electrocautery was applied to the interior of the lesion and a thorough curettage was performed (Fig. ).\nThe lesions removed by cyst enucleation were sent for tissue biopsy. The largest lesion was approximately 3 × 2 × 1.5 cm and was lined by a thick, fibrotic tissue layer (Fig. ).\nHematoxylin and eosin (H and E) stained sections showed dense collagenous tissue surrounding the osseous trabeculae, and peritrabecular clefting was present (Fig. a). Mitosis or atypia was not seen (Fig. b). The biopsy result revealed active nonspecific chronic inflammation with fibrosis.\nIn view of the radiographical and intraoperative findings, the absence of any history of trauma, a low probability of dental infection being the cause (as per endodontic and periodontal examinations), absence of evidence of malignant transformation (confirmed by histological findings) [–], the researchers confirmed that the case involved non-specific cystic degeneration in the CFD site. Postoperative healing was uneventful (Figs. and c), as was the postsurgical follow-up over a 6-month period (Figs. and d). Thereafter, the patient was lost to follow-up. |
A 20-year-old female with a long history of complex borderline personality disorder, post-traumatic stress disorder and factitious disorder characterized by multiple admissions for overdoses and foreign body ingestions presented to the emergency room following ingestion of multiple 2-cm finishing nails, dispersed throughout the small bowel on abdominal X-ray (). She was admitted for observation but on the fourth day left against medical advice (AMA) and returned after swallowing additional nails and one magnet. This magnet measured radiographically with a dimeter of 2 cm and height of 0.75 cm. She was taken for an esophagogastroduodenoscopy using a pediatric gastroscope but the magnet had already passed beyond the proximal jejunum (). Three days later she left AMA for the second time and returned having overdosed on acetaminophen and swallowed additional magnets. Two days afterwards, following medical clearance by internal medicine, the X-ray in was taken showing an additional four magnets aggregated with nails and the first magnet in the right lower quadrant. Serial X-rays demonstrated no movement of this complex. She remained stable with intermittent abdominal pain but no clinical or radiographical signs of bowel obstruction. Six days later, a computer tomography scan of the abdomen showed this complex to be lodged within the terminal ileum ().\nAfter another 8 days without movement the patient was prepared for a colonoscopy. A large conglomerate of nails was extracted from the bowel wall of the ileocecal valve with endoscopic forceps and snare () and placed in the cecum (). Some of these nails appeared to be extracted through the wall of the cecum, raising the possibly of an enteric-colonic fistula formed by impaction of the mass. Intubation of the terminal ileum did not show any retrievable objects. A Roth Net® retrieval basket was used to extract a total of 10 collected nails from the cecum (). Subsequent X-rays over the next several days showed migration of the remaining objects in colon () which were subsequently eliminated.\nUnfortunately, despite multiple psychiatric interventions, this patient continued overdosing and ingesting foreign bodies, which would ultimately lead to her death 2.5 years later. |
The second patient died was a 39-year-old female with univentricular heart (tricuspid atresia), who had had previously two cardiac surgeries (the last was atrio-pulmonary Fontan, 33 years earlier). The patient suffered from a serious right atriomegaly with frequent episodes of atrial tachycardia, so she was a candidate for Fontan conversion surgery. Pre-operative ventricular function was mildly reduced (50%). In the post-operative, the patient suffered from severe single ventricle dysfunction resulting in LCOS and the need for ECMO implantation. Both patients could not be weaned from ECMO due to multi-organ failure.\nSeven patients required pace-maker implantation due to post-operative sinus node dysfunction or atrioventricular conduction abnormalities. All patients were discharged on oral antiarrhythmic for 3–6 months and anticoagulants for 6 months. At discharge, 15 patients were in sinus rhythm, 5 had a stable pacemaker rhythm, 2 had atrial fibrillation, and 1 atrial flutter. One patient discharged in sinus rhythm had a pacemaker implant 5 months after the operation due to the presence of sinus node dysfunction.\nDuring a median follow-up of 14 months (IQR 7–27), there was no late mortality and 17/23 patients had an improvement of NYHA functional class. Five patients in NYHA III progressed to class II and 4 to class I; eight patients progressed from class II to class I.\nAt follow up electrocardiogram, 16 patients were in sinus rhythm, 6 with stable pacemaker rhythm, and 1 with permanent atrial fibrillation.\nRecurrence of arrhythmia occurred in 2/23 (8.6%) patients, more than 3 months after surgery. These patients presented at surgical ablation with history of atrial fibrillation lasting 4 and 19 years, respectively, and both had atrial fibrillation, which was treated with right-sided Maze rather than Cox maze III due to technical issues. Sixteen (69%) patients are in stable sinus rhythm, 12 without any anti-arrhythmic therapy. At median follow up of 14 months (IQR 7–27), freedom from recurrence of arrhythmia was 90.9% and cumulative risk of recurrence was 9.6% (). |
A 53-year-old dark-skinned male, who has provided consent for his case to be published, presented to establish care with nephrology for reported chronic kidney disease and hypertension. Baseline renal and bladder ultrasound were obtained and revealed a large pelvic mass for which he was referred to urology.\nThe patient denied any systemic symptoms. His only urologic complaint was erectile dysfunction that responded well to phosphodiesterase inhibitors (PDEIs). At an outside institution 10 years ago, he underwent right orchiectomy and left orchiopexy for bilateral undescended testicles. However, the patient's path report read “connective tissue without atypia” without mention of a testicle.\nGenitourinary examination was consistent with a virilized male []. Pertinent findings included an empty scrotal sac, bilateral inguinal scars, a small palpable gonad in the distal left inguinal canal, and a 20cc smooth prostate gland.\nNoncontrast computed tomography of the abdomen and pelvis revealed an 11 cm complex pelvic mass that appeared to originate near the right seminal vesicle []. Given, the unclear history of the right orchiectomy, testicular malignancy was of significant concern. Chest X-ray and serum testicular tumor markers were normal.\nWe discussed with the patient that we still lacked a definitive diagnosis and could not yet determine the malignant potential of the radiologic finding. Because of a recent divorce and separation from his daughter, he was homeless but currently residing in a nearby mission. Given his unsteady social situation, all involved parties felt it best to proceed with exploratory surgery.\nIntra-operatively, a uterus was discovered with the evidence of a gonad situated near the right fallopian tube []. The right gonad was removed due to inadequate vascular length to bring it to the scrotum. Because the pelvic mass was the primary source of his anxiety, gynecology was consulted intra-operatively for assistance with resection of the Müllerian structures, taking particular care not to disturb the blood flow to the remaining left gonad. Given his good preoperative erectile function on PDEIs, the dissection was carried just short of the prostate.\nFinal pathology confirmed that the right gonad was indeed the right testicle with an attached vas deferens, and the remaining organs were consistent with bilateral fallopian tubes and uterus with fibroids. A karyotype was sent revealing 46, XY. |
A lean 15-year-old white Caucasian female was diagnosed with type 1 diabetes. At the time of diagnosis, the patient presented with polyuria, polydipsia and a random glucose of 20 mmol/l. She tested positive for anti-gliadin antibodies and islet cell antibodies. She had no significant illness prior to this and there was no family history of diabetes or any other autoimmune diseases. Examination found no features of insulin resistance. She had good glycaemic control during the first 6 months following diagnosis. Her plasma HbA1c was maintained at 58 mmol/mol with s.c. Mixtard 30, in a dose of 32 units twice daily, which was increased to four times a day within two months of diagnosis. She had never received animal-derived insulin preparation.\nAfter 8 months from the time of diagnosis, the patient reported cyclical swings in her insulin requirements according to her menstrual cycle. Her insulin requirements in the first 2 weeks of the cycle were around 60 units/day. This was followed by a week where the insulin requirement rose to 90–120 units/day, and within 4 months her insulin requirements were 280 units/day, with little effect on blood glucose levels. This period was usually followed by a week when she required no insulin to be administered (average glucose of 3.2 mmol/l).\nAfter 2 months, the cyclical swings of insulin became worse. Her insulin requirements continued to rise up to 300 U/day. These periods were usually preceded by intervals of hyperglycaemia and ketosis that resulted in repeated admission to the intensive care unit, where the patient was managed with i.v. insulin. Furthermore, the period when insulin was not required became shorter and was followed by long periods of hypoglycaemia. The hypoglycaemic episodes were very severe, with un-recordable glucose concentrations, and the patient required continuous infusions with 15–20% dextrose at an infusion rate of 200 ml/h for up to 2 weeks. Any interruption in i.v. infusion of dextrose led to the immediate recurrence of hypoglycaemia. These episodes became recurrent and nocturnal. Surreptitious insulin self-administration or insulin administration by proxy was considered but no evidence was found. |
A 53-year-old man presented with a gradually increasing mass located in the soft tissue of the oral cavity. The lesion was biopsied, and the pathology report showed increased infiltration of the buccal mucosa with monoclonal plasma cells. Immunochemistry demonstrated positivity for CD138, CIgA, and negativity for CD56, Cyclin D1 and CD20. Therefore, the diagnosis of soft tissue plasmacytoma was confirmed, for which he received localized radiotherapy with a total dose of 40 Gy, due to the absence of systemic disease as the bone marrow biopsy revealed the absence of neoplastic infiltration and serum and urine immunofixation were all negative.\nAfter five years, the patient noticed a painless swelling in his right testis. An ultrasound of the scrotum was performed that showed a hypoechoic mass in the right testicle with increased vascularization, and a normal-appearing left testis (Figure ). The patient subsequently underwent a right radical orchiectomy and the histopathology report showed testicular infiltration by a plasma cell neoplasm with identical immunophenotype (CD138+, CIgA+, CD56-, CD20-, Cyclin D1-) to the primary site in the oral cavity. Subsequently, the patient underwent a bone marrow biopsy that showed the absence of monoclonal plasma cell infiltration, and a PET/CT scan that was negative for reactive lesions suspicious of malignancy. In addition, serum and urine protein electrophoresis, as well as serum-free light chain assay were all within normal range, thus, excluding the presence of systemic disease, while complete blood count and full biochemical profile were normal.\nHowever, after one year, the patient presented again with painless swelling of his left testis that was attributed to disease relapse based on the imaging findings (Figure ). He refused to undergo left orchiectomy, thus he proceeded with systemic treatment based on lenalidomide, bortezomib and dexamethasone. Following 4 cycles of treatment, the patient underwent high dose melphalan with autologous stem cell transplantation. The blood tests of the patient revealed an increase in serum creatinine (1.3 mg/dl with 1mg/dl baseline level), normal calcium levels and normal complete blood count. Complete response was achieved with negative PET/CT scan and negative marrow minimal residual disease assessment. The patient continued on lenalidomide maintenance; however, six months later he was diagnosed with disease relapse in his left testicle (Figure ). A PET/CT scan was then performed, and showed a reactive lesion with an increased SUV max of 7 in the left testis, with no other loci suspicious of disease relapse. The blood analysis of the patient revealed no abnormal values. Since the patient refused surgery, he was initiated with next-line therapy, with bortezomib, cyclophosphamide and dexamethasone, he responded partially and still remains in remission (Figure ), while serum protein electrophoresis, immunofixation and serum-free light chain assay, all remain within normal levels. |
The proband was a 35-year-old male from Hebei province in the north of China. He is of Han ethnicity and was born to consanguineous parents. His family pedigree is shown in Fig. . The proband was normally delivered after a full-term pregnancy, and birth weight and length were within normal ranges. The initial signs and symptoms appeared when he was 6 years old. Deformity of interphalangeal joints initially appeared in the fingers. Hips, knees, and wrists were then gradually involved. Diagnosis of JRA was considered by local hospitals, and glucocorticoids were prescribed without any efficacy. As he grew up, his symptoms deteriorated. He had to walk with crutches at 16 years of age because of arthritis of the lower extremities. At 26 years of age, he first experienced progressive pain with numbness radiating down his entire left leg and right thigh. At 34, he started to have mild difficulty in urination. Thereafter, his leg pain progressed and he became immobile. Treatment with tramadol, physical therapy, and spine injection were tried but were not effective. He had a younger brother with a similar clinical presentation but who also had mild neurological impairment (Fig. ).\nThe proband’s height and weight were 162 cm and 72.5 kg when he was admitted to our hospital. His visual analogue scale (VAS) score was 9. He did not have behavioral difficulties and was not retarded in his intellectual development. Physical examination showed multiple malformations of the major limb joints, especially of the knees and hands (Fig. ). Amyotrophy of both lower limbs was obvious. Cervical and lumbar movements were limited with compensatory kyphosis. The muscular strength of all four limbs was normal. Dysesthesia was found in the posterolateral left calf, dorsolateral left foot, and perineal area. Bilateral knee-jerk reflexes and ankle reflexes were hypo-induced. The erythrocyte sedimentation rate (13 mm/h) and C-reactive protein level (2 mg/L) were both within the normal range. Tests for rheumatoid factors were negative.\nSpinal x-rays showed flat and osteoporotic vertebral bodies. Pedicles were short, and end plates were irregular. Bone bridges were seen at many levels. Kyphosis was detected in both the cervical and upper thoracic spine. Magnetic resonance imaging showed multilevel Schmorl nodes. Multilevel disc herniation and hypertrophic ligamentum flavum caused lumbar canal stenosis from L2 to S1 (Fig. ). |
We present a primigravid, 33-year old European woman, who was admitted to the emergency department for obstetrics in her 34th week of pregnancy, due to a symptomatic but compensated anemia, which clinically has been manifesting in a 3-week history of fatigue, mild circulatory insufficiency and a hemoglobin level of 4.8 g/dL. The patient negated vaginal or rectal bleedings but mentioned a blackening of stool during the last three weeks, which she attributed to the intake of iron supplements. There were no abnormal findings considering the pregnancy. After transfusion of three packed red blood cells, only a marginal increase in hemoglobin level (5.9 g/dL) was recorded. Considering the symptoms and insufficient increase in hemoglobin levels, a gastrointestinal bleeding was assumed and gastrointestinal endoscopy was advised. Gastroscopy revealed a posterior wall-sided mobile conglomerate tumor in the corpus and antrum with an ulcerated and hypervascularized surface leading to severe hemorrhage. Proximal to the tumor, the posterior wall of the gastric corpus was impressed extraluminally. According to its endoscopic appearance, the tumorous mass was assumed as a highly suspicious finding with the suggestion of an ulcerated malignant gastric tumor or gastrointestinal stromal tumor (GIST) of the stomach. Biopsy or intervention measures were not performed due to the risk of further uncontrollable bleeding. The extent of the tumor was additionally determined by transabdominal ultrasound, which showed an 11 cm mostly cystic tumor mass presumably arising from the pancreatic tissue. At this point, the entity was still not clear. An interdisciplinary team of gynecologists, visceral surgeons and gastroenterologists evaluated these clinical findings with regard to the gestational age and decided to refrain from further diagnostics (magnetic resonance imaging (MRI)/computed tomography (CT)/biopsy/blood tests) and conservative therapy because of the urgent need of maternal treatment. A preterm delivery via cesarean section was indicated and followed by visceral surgery in the same session. Intraoperatively, a differentiation between a conglomerate tumor arising from the gastric posterior wall or the pancreas was impossible (). A tumor resection was performed, while preserving a small gastric pouch and the post-pyloric duodenum. A distal pancreatectomy and, as the splenic vein seemed to be infiltrated as well, a splenectomy with resection of the splenic vessels followed. Finally, a reconstruction of the gastrointestinal tract analogical to commonly performed bariatric surgeries, speaking of a Roux-en-Y reconstruction with gastrojejunostomy, completed the surgery. Macroscopically, there were no signs of intraabdominal metastases. The postoperative recovery remained without complications. The premature eutrophic infant (1900 g, APGAR 7/8/9, pH 7.36) only had a short adaption disorder and CPAP (continuous positive airway pressure) therapy in the first minutes of life. There were no further major events in the treatment of the newborn.\nPathology confirmed a 12.5 cm × 11 cm × 10.5 cm measuring, mostly cystic, partly necrotic, knotty pre-bulged tumor, which was located on the posterior gastric wall and was removed with tumor-free resection margins (). The spleen and twenty-five lymph nodes were not affected. Multi-chambered cystic structures, lined by a mucin-producing columnar epithelial layer and ovarian-type stroma consisting of spindle-shaped cells with round or elongated nuclei and sparse cytoplasm, were observed through light microscopy (A,B). Further immunohistochemical staining confirmed the expression of progesterone receptors (PR) and a negativity for estrogen receptors (ER) in the ovarian-type stroma (C,D). Thus, unexpectedly, the final diagnosis of a pancreatic mucinous cystic neoplasm (MCN) with low grade dysplasia was made. More specifically, no evidence of malignancy was found. |
This is a 30-year-old man who was well until the age of 13 years when he started tripping and falling. He was observed to be dragging his feet and developed a clumsy gait. These symptoms progressed and he started to use a cane at the age of 16, a walker at the age of 18, and, ultimately, a wheelchair at the age of 24 years. During this time period he had numerous neurological evaluations and was diagnosed with spastic paraparesis. He has had no associated complaints of pain, numbness, bowel or bladder symptoms, seizures, visual abnormalities, or vertigo. In the last few years, he has developed symptoms of dysarthric speech and clumsiness of his upper extremities. He was diagnosed with attention deficit disorder and further evaluations showed IQ scores (Wechsler Intelligence Scale for Children) of 107 (verbal), 100 (performance), with an overall score of 103. He went on to complete high school and postsecondary education.\nHe is the product of a normal full term pregnancy and although both parents are of Italian origin, there is no consanguinity. There is no family history of any neurological disorder on either the paternal or maternal branches of the family. The patient has no siblings.\nGeneral physical examination revealed no abnormalities. Neurological examination performed showed a minimental status score of 30/30. His speech was dysarthric but otherwise the cranial nerve and sensory examinations were normal. The stretch reflexes were diffusely pathologically brisk and he had sustained clonus at the ankles with bilateral extensor responses. Motor examination revealed a marked hypertonia across all joints bilaterally. Both lower extremities were extended at the knee joint and very spastic. Power was full in the upper extremities (MRC Grade 5/5) and reduced in the lower extremities (MRC Grade 3/5 distally). He could not stand unassisted. A neurological examination was performed on both parents and revealed no abnormalities.\nDuring the course of his medical care many investigations were performed and the following tests were either normal or negative: routine serum chemistries, cell count and differential, creatine phosphokinase, renal and thyroid function studies, and serology for HTLV1.\nIn addition, magnetic resonance imaging studies of his spinal cord and brain were normal. An electromyography indicated no evidence of neuropathy or myopathy.\nA lipid profile of the patient had been performed as part of routine screening and was abnormal. Subsequently, lipid profiles of both parents were performed (). |
A 67-year-old male with a past medical history of congestive heart failure, non-ischemic dilated cardiomyopathy, atrial fibrillation, and pacer-dependent heart block, presented to the University of Miami emergency department with refractory complex partial seizures. Due to the fact that the patient was pacemaker-dependent, he was unable to undergo MRI. Therefore, a contrasted CT scan of the brain was performed, which demonstrated a heterogeneously enhancing left frontal mass just anterior to the left motor cortex thought to be either a primary central nervous system tumor or a metastatic lesion (Figure ).\nA full metastatic workup was conducted with no primary lesion detected. Given the location of the lesion, further imaging was thought to be necessary for two reasons: 1) to further elucidate the differential diagnosis of the lesion and 2) to help safely guide resection or biopsy of a lesion in close proximity to eloquent brain tissue. As a result, cardiothoracic surgery was consulted for the possible replacement of the patient’s current pacemaker with an MRI-compatible alternative. The patient subsequently underwent the removal of their pacemaker and replacement with a Surescan Medtronic DDD Pacemaker RVDR01 (Minneapolis, MN, US). Following this procedure, an MRI was performed and demonstrated a multi-lobulated, relatively uniformly enhancing left frontal lesion (Figure ). Due to the lack of a definitive diagnosis, the decision was made to perform an awake craniotomy with MRI-assisted stereotactic guidance and intraoperative electrocorticoraphy for a definitive diagnosis. The lesion was able to be resected with the initial pathology suggestive of Mycobacterium tuberculosae brain abscess. The patient was placed on anti-tuberculosis medication and intravenous (IV) antibiotics and discharged home. Approximately one year later, he returned to the hospital after suffering a partial seizure with subsequent right upper extremity weakness. He was found to have a recurrence of his left frontal lesion and was again taken to the operating room for surgical resection. Repeat imaging at one year did not demonstrate any further recurrence of his previously seen lesion. |
A 51-year-old man with a history of chronic HBV infection for more than 30 years presented to the Hepatic Surgery Center (Tongji Hospital, TongjiMedical College, Huazhong University of Science and Technology, Wuhan, China) with abdominal discomfort in September 2015. Enhanced abdominal magnetic resonance imaging (MRI) revealed a mass with the largest diameter measuring up to 11.0*9.5cm in size in segments 7 and 8 within the right lobe of the liver ( and ). The patient was diagnosed with HCC with the background of cirrhosis secondary to HBV infection based on imaging studies and confirmed with the clinical diagnosis of Barcelona Clinic Liver Cancer (BCLC) A and Child-Pugh class A. Entecavir treatment was routinely used once per day from that time. His alpha-fetoprotein (AFP) level was in the normal range. On September 23, 2015, he underwent segment 7 and 8 liver resection and cholecystectomy. The postoperative pathological examination showed hemorrhage with necrosis in the middle of the tumor, with moderate differentiation and vascular cancer embolus. The incisal edge was negative and did not invade the hepatic capsule ( and ). He recovered well and discharged from hospital. With regular examination, unfortunately in March, 2017, he had liver mass recurrence and lung metastases (BCLC C). According to the guidelines, he started systemic treatment with sorafenib (400 mg, twice per day). Enhanced abdominal computerized tomography (CT) scan showed a heterogeneous irregular mass measuring up to 2.3*2.2cm with arterial phase enhancement and venous phase washout in the left lobe of liver (). Chest CT scan revealed that multiple pulmonary nodules on both sides of the lung which were diagnosed as lung metastases, the largest up to 8-mm in diameter ( and ). The patient received percutaneous microwave coagulation for the liver tumor on March 23, 2017. On April 5, 2017, he proceeded with TACE and then pulmonary arterial infusion (PAI) on December 27, 2017. On December 24, 2018, enhanced abdominal CT scan showed two lesions with the largest diameter greater than 3 cm in both sides of the liver (–). He had radiographic progression in lung metastases after 3 months ( and ). Disease progression was measured using Response Evaluation Criteria in Solid Tumors version 1.1 (RECIST v 1.1) or modified RECIST for HCC (mRECIST). Under this circumstance, he received 160 mg regorafenib orally once daily for 3 weeks in each 4-week cycle. The patient had no dose reductions in the period of medical treatment. On June 12, 2019, restaging chest CT scan showed partial response (PR) in lung metastases ( and ). Despite good tolerability of regorafenib, repeat MRI scans of both chest and abdominal on December 3, 2019 revealed obvious tumor progression in liver and lung ( and and and ). The patient started treatment with regorafenib 160 mg in combination with sintilimab (PD-1 inhibitor) on December 4, 2019. Sintilimab was given 200 mg over a period of 30–60 minutes for every 3 weeks as a cycle. The patient tolerated well the treatment, except potentially treatment associated general pruritus grade 2, with mild skin changes occurring but without rash. After five cycles of sintilimab injection, a follow-up abdominal MRI scan showed complete response (CR) in target lesions of liver without any tumor activity, as assessed by mRECIST ( and ), while chest CT scan revealed CR in target lesions of lung, as evaluated by RECIST v 1.1 ( and ). showed local and systemic treatment and a summary diameter of target lesions according to RECIST v 1.1 or mRECIST criteria. During the process of treatment, the patient had no severe complications and showed good liver functions (Child-Pugh A) (). At his last follow-up, nearly 5 years have elapsed since the diagnosis of HCC, and it is up to 40 months since lung metastases have been diagnosed. He was in a very good condition, without evidence of disease progression till now. Written informed consent was obtained from the patient for publication of this case report and any accompanying images. This study was approved to publish the case details by the Ethics Committee of Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China. |
A 41-year-old housewife initially presented to the Cardiology Department of Hedi Chaker Hospital in Tunisia in November 2011 with symptoms of exercise intolerance and occasional palpitations of several months’ duration. She had no family history of congenital defects. Three years earlier, when she was 38 years old, she gave birth to a normal baby after undergoing an uneventful full-term pregnancy and delivery.\nCyanosis and dyspnoea on exertion had been present throughout her life, but despite this, she appeared to have had a fairly normal life, being able to tolerate daily activities well. Two weeks prior to admission, she reported having experienced an exacerbation of dyspnoea.\nShe was a small-built woman and appeared deeply cyanosed on examination but not dyspnoeic at rest. Prominent clubbing of the fingers was noticeable. Congestion of the jugular veins was also striking. Her oxygen saturation in room air was about 80%. Physical examination revealed a mild systolic murmur over the left parasternal border and a loud second heart sound in the right second intercostal space.\nA 12-lead resting electrocardiogram revealed sinus rhythm, right bundle branch block and high QRS voltage suggestive of biventricular hypertrophy. A chest radiograph revealed marked cardiomegaly with a prominent main pulmonary trunk and increased pulmonary vascularity.\nTransthoracic echocardiography indicated a levocardia heart with atrial situs solitus and concordant atrioventricular connections. Marked biventricular hypertrophy in the fourchamber view was also evident. The left ventricle demonstrated a normal ejection fraction. The most striking finding was a single large vessel arising from the base of the heart, with mild regurgitation related predominantly to the summit of the right ventricle (70%). A large, non-restrictive outlet VSD was noted beneath the truncal valve (). Neither the pulmonary artery (PA) nor the pulmonary valve could be seen.\nCardiac MRI was also performed to better delineate the origin of the pulmonary arteries. It demonstrated a dilated common arterial trunk with the left and right pulmonary arteries arising from a short main pulmonary trunk at the posterior side of the common arterial trunk. The left ventricle (LV) was normal in size. The right ventricle (RV) was also normal in size with concentric hypertrophy. A large, subarterial VSD was noted beneath the truncal valve, which was trileaflet, with mild insufficiency ().\nFrom on the above findings, for this patient, a diagnosis in keeping with a conotruncal anomaly could best be classified as type 1, based on Collette and Edwards’ classification. Furthermore, the thoracic vasculature was significantly altered, with irreversible pulmonary hypertension. Taking this into account, conservative management was recommended.\nThe patient appeared to make good progress on medical treatment, which included bed rest and fluid restriction. She was advised against pregnancy, considering her mature age and in light of the underlying severe pulmonary hypertension. Her condition appeared to have remained stable 15 months after discharge from hospital. Chromosomal studies were not undertaken, however her child appeared in a good state of health. |
The second case shows a 53-year-old female. Clinical and radiographic examination confirmed a unilateral posterior crossbite due a transverse maxillary deficiency with a significant mandibular skeletal deviation towards the side of the crossbite (Fig. ). Treatment objectives.\nSARME was planned to correct the transverse discrepancy followed by arch leveling with lingual appliances and then a second surgery to correct the mandibular asymmetry.\nSimilar to case 1 impressions were obtained and this time the lingual appliances were manufactured by DW Lingual Systems (Bad Essen, Germany).\nDuring the planning for the production of the lingual brackets, it was noted -similar to case 1- that a surgically assisted rapid maxillary expansion takes place. The transverse width of the upper jaw should be adapted to the lower jaw.\nTwo trans sagittal Benefit mini-implants were inserted in the T-Zone. A silicon impression with the transfer caps was taken. The impression was given to the laboratory together with the lingual molar bands. A Hybrid Hyrax [] was then made and laser welded to the molar bands (Fig. ). Similar to case 1, the lingual appliance was indirectly bonded with a dual cured resin and the maxillary expansion appliance was inserted. In this case the molar bands were cemented with a dual cured resin and the hybrid hyrax was fixed to the mini-implants using the Benefit fixation screws. The first lower arch wire 12 NiTi was inserted while in the upper the brackets were secured with a continuous steel ligature in each quadrant (Fig. ). SARME was performed with an activation rate of two quarter turns twice a day until crossbite correction was achieved at two weeks post-surgery (Fig. ). The Hybrid Hyrax was then blocked. The first upper archwire (12 NiTi) was placed four weeks after surgery (Fig. ). After complete leveling and radiographic re-examination the surgery to correct the asymmetry was performed.\nThe patient has a positive overbite and overjet now. The patient shows a good transversal and sagittal occlusion. |
A 43-year-old male from Guatemala presented as a direct admission for untreated HIV complicated by cytomegalovirus (CMV) ileitis, 40 lb weight loss over one year, worsening abdominal pain, and odynophagia. His medical history was notable for untreated HIV diagnosed six years prior to admission and a remote history of methamphetamine abuse.\nTwo months prior to his hospitalization, the patient was hospitalized at an outside hospital in California for abdominal pain and a 40 lb weight loss. The initial CT abdomen showed an SBO and thickening in the terminal ileum, concerning inflammatory bowel disease (IBD). An esophagogastroduodenoscopy (EGD) at that time was unremarkable though biopsies were obtained. A colonoscopy demonstrated ulcers in the terminal ileum, and the patient was ultimately discharged on a two-week course of prednisone 40 mg with concern for new onset of Crohn’s disease. The biopsy pathologies subsequently returned with intraepithelial lymphocytosis on the duodenal tissue biopsy, scattered CMV inclusions by immunochemistry on the terminal ileum tissue biopsy, and negative for IBD, but these pathology reports, unfortunately, resulted after the patient had been discharged and he was unable to be reached to initiate therapy. There were no stains performed for histoplasmosis specifically, although the reasons are unclear.\nTwo months later, he was again admitted to the hospital for worsening abdominal pain. Upon admission, the patient stated that he had been having diffuse abdominal pain and intermittent non-bloody diarrhea for two months, as well as weight loss of 40 lbs over the past year. He had also experienced persistent throat pain with swallowing for the past month and had been eating significantly less due to the odynophagia. He denied dysphagia to solids and liquids, acid reflux, fever, chills, headache, joint pains, and new rashes. He stated that he had not taken any anti-HIV medications since diagnosis six years ago.\nOn initial physical examination, vital signs were as follows: afebrile, blood pressure (BP) 114/86, heart rate (HR) 99, respiratory rate (RR) 18, SpO2 100% on RA, and BMI 26. The physical exam was unremarkable.\nThe patient’s labs were notable for the following: Creatinine (Cr) 0.63 mg/dL, white blood cells (WBC) 3.8 10e9/L, Hgb 10 g/dL, MCV 77.5 fl, Platelet 341 10e9/L, absolute lymphocyte 0.7 10e9/L, CD4 47, HIV viral load 150,000, alkaline phosphatase 279 IU/L, AST 67 U/L.\nHe stopped smoking 30+ years ago, denied alcohol use, and current use of methamphetamine, and stated that he was currently only sexually active with his wife. He moved to the United States from Guatemala 22 years ago. He lived in California for 15 years and then lived in Minnesota for the past seven years, and has been living at his friend’s home in the suburbs of Minnesota. Due to his undocumented status, he has never worked. He denied any known outdoor exposures to Histoplasma in Guatemala and it is unclear if he has had exposures to Histoplasma since moving to the United States.\nDue to his ongoing odynophagia, an EGD was again performed with a biopsy obtained that showed non-specific reactive changes in the squamous mucosa of the esophagus and mildly increased intraepithelial lymphocytes with normal villous architecture in the duodenum. Ganciclovir was started for untreated CMV ileitis.\nA chest X-ray was obtained for a cough, and it demonstrated a diffuse miliary pattern, with subsequent CT chest demonstrating extensive miliary nodules bilaterally with an upper lobe predominance (Figure ). Subsequent urine Histoplasma antigen was positive. Also, after three days post collection, Histoplasma grew from the right upper lung lobe bronchoalveolar lavage (BAL) fungal culture. Fungal elements consistent with Histoplasma capsulatum, per pathology, were found on microscopy, and identification was also confirmed with polymerase chain reaction (PCR) test. Aerobic and anaerobic blood cultures were negative, and no fungal blood cultures were obtained.\nSeven days after admission, the patient developed worsening abdominal pain and a CT abdomen/pelvis at that time demonstrated markedly dilated distended loops of small bowel with decompressed loops of ileum and colon consistent with a high-grade SBO. Subsequent magnetic resonance (MR) enterography abdomen/pelvis showed evidence of long segment active enteritis involving much of the distal ileum with narrowing in the distal ileum in the right mid-abdomen suspicious for a stricture and ongoing colitis in the ascending colon (Figure ). The patient underwent a laparoscopic-assisted ileocecectomy and ileocolic anastomosis, and tissue biopsy demonstrated histoplasmosis-associated enteritis in the terminal ileum (Figure ). The Grocott-Gomori’s methenamine silver (GMS) stain of the terminal ileum highlighted abundant fungal organisms, morphologically consistent with Histoplasma capsulatum. CMV stain of the colon (terminal ileum, cecum, appendix) was negative for CMV inclusions.\nThe patient was ultimately started on antiretroviral therapy (bictegravir, emtricitabine, and tenofovir alafenamide) for his previously untreated HIV. For his disseminated histoplasmosis, the patient was treated with amphotericin B intravenously (IV) for four consecutive weeks and was transitioned to voriconazole, rather than itraconazole with the patient’s transaminitis, for 12 months.\nPrior to discharge, the patient decided to move back to his home state. The infectious disease (ID) physicians contacted the patient’s home primary care physician and his ID team prior to discharge for continuity of care. Due to the patient’s move outside of our healthcare system, no follow-up about his treatment progress is available. |
A 55-year-old female, P3003, presented with postmenopausal spotting on and off for the past 6 months, cough with expectoration, and significant weight loss for past 2 months. For these complaints, she had been put on empirical antitubercular drugs at some outside hospital. She took these medications for 2 months but had no improvement in her chest condition and rather the bleeding episodes and pain in lower abdomen increased significantly. She was then referred to our center. The patient was nondiabetic and nonhypertensive, and there was no history of long-term exposure to estrogen. She was postmenopausal for 3 years. On examination, her general condition was poor. She was cachexic with weight 27 kg and body mass index of 15 kg/m2. On auscultation, bilateral air entry was decreased and rhonchi were present. Abdominal examination revealed a scaphoid abdomen and a lump corresponding to 14 weeks size gravid uterus, hard in consistency, and restricted mobility in the suprapubic region. On per speculum examination, the cervix could not be visualized as it was pointing acutely forward. Hence, cervical screening and endometrial biopsy could not be done. On bimanual examination, a hard mass of around 14 weeks was felt, which was nonmobile and nontender.\nHer hemoglobin was 7.1 g%. Rest all investigations were within normal limits. Ultrasonography showed evidence of a well-defined heterogenous hypoechoic mass lesion 20 mm × 17 mm in part of the myometrium of the fundal region likely intramural fibroid and bulky uterus. However, the magnetic resonance imaging (MRI) report was suggestive of endometrial carcinoma involving more than half of the myometrium. Computed tomography (CT) of the chest was advised to rule out metastasis. However the patient was lost to follow-up for about 2 weeks when she returned back with CT report suggestive of traction bronchiectasis and discrete random nodules with feeding vessel signs in the right lower lobe of lung, likely metastases. Surprisingly, there was enlargement of uterine mass to approximately 20 weeks size in 2 weeks duration with increase in pain abdomen and fall in hemoglobin to 5.2 g %. A repeat ultrasonography was done which showed a large lobulated mass lesion with heterogeneous echotexture, size 13.7 cm × 7.3 cm with increased vascularity on Doppler. The uterus with bilateral adnexa could not be separately seen.\nAt the same time, the patient complained of severe pain in the left leg with unilateral edema. Homan's sign was negative. Doppler of the leg was normal. Hence, deep vein thrombosis was excluded. Seeing the rapid growth of mass and falling hemoglobin, a working diagnosis of leiomyosarcoma was made. After receiving two units of packed red blood cells, the patient was posted for exploratory laparotomy.\nIntraoperatively, the mass was up to the umbilicus, adherent to the omentum and sigmoid colon. It had actually ruptured inside the abdomen with tumor tissue and necrosed material adherent to the bowel, abdominal wall, and lateral pelvic wall []. Adhesiolysis was done. Total abdominal hysterectomy with bilateral salpingo-oophorectomy and infracolic omentectomy was done. The specimen was sent for histopathology.\nThe resected specimen measured 13 cm × 13 cm × 4 cm. The outer surface was congested with multiple surface deposits. Cut section showed dilated endometrial and endocervical cavity. Endometrial cavity was filled with friable gray–brown growth []. Multiple small growths were also seen in the myometrial cavity.\nTumor was arranged in sheets and lobules and separated by fibrous septae of varying thickness. Individual tumor cells were small to medium sized, round having round to irregularly contoured, vesicular nuclei, small nucleoli, and scant to moderate amounts of eosinophilic to granular cytoplasm. Brisk mitotic activity is seen. Several bizarre cells and a few multinucleated tumor giant cells and large areas of necrosis were seen. Hemorrhage, fibrinous deposits, and several lymphovascular emboli were seen. Morphologically features were suggestive of HGESS []. The left ovary and the left fallopian tube also showed metastatic tumor deposits with lymphovascular emboli. The tumor involved the muscularis propria of the appendix.\nImmunohistochemistry was positive for CD 10 [] and negative for estrogen and progesterone receptors.\nThe patient received three units of packed cells, eight units of fresh frozen plasma, and four units of platelet intraoperatively because of massive blood loss and subsequent hypotension. There was generalized oozing from all the sites. Hence, the pelvis was packed to give a tamponade effect and abdomen closed. She was kept on ventilator support in the intensive care unit. After 48 h, we reopened the abdomen to remove the pack. There was no bleeding, but she had developed ascites. A drain was kept in situ. In the postoperative period, the patient was conscious and responsive but could not be extubated due to her poor chest condition. She developed herpes zoster on the 4th postoperative day and that justified the pain that she had preoperatively to be preherpetic neuralgia. Intravenous acyclovir was given for 7 days.\nExtubation was tried many times, but she failed to maintain without ventilatory support. Bronchial aspirate revealed the presence of Acinetobacter species for which cefoperazone-sulbactam and colistin were given for 8 days. To wean her off the ventilator, tracheostomy was done. However, despite all efforts, patient expired on day 28 of operation. |
A 59-year-old man presented in primary care with urinary symptoms and vague pelvic pain. His GP arranged for CT scan of the abdomen and pelvis and a PSA blood test. He was then referred to our centre following the finding of a raised PSA (9.3mcg/L). The patient suffered from mixed mild LUTS with an IPSS of 7 and QoL of 3. There was no history of UTI or visible haematuria. His past medical history included left inguinal hernia repair associated with left simple orchidectomy for atrophic testicle, at 11 years of age.\nAbdominal examination revealed the scar related to left inguinal hernia repair. Digital rectal examination revealed a firm right prostate base.\nThe patient then underwent a multi-parametric MRI (mpMRI) scan of the prostate (T2W, DW and Dynamic Contrast Enhanced sequences) prior to transperineal prostate biopsy.\nHis CT and mpMR imaging together with biopsy findings, were then discussed in our multidisciplinary team (MDT) meeting. CT urogram () showed a horseshoe kidney with atrophic left part and a supernumerary right kidney. The ureter of the atrophic left horseshoe kidney was draining into the ipsilateral seminal vesicle whereas both the right horseshoe and the supernumerary right kidneys drained via a single ureter into the urinary bladder. MpMR images () showed a 26 cc prostate with a PI-RADS 4/5 lesion at the right posterior peripheral zone at the base. They also showed an abnormally enlarged left seminal vesicle. His prostate biopsies confirmed right side Gleason 3 + 4 in a significant number of cores. Consensus from MDT meeting was that the patient should be offered active treatment options for his clinically significant prostate cancer. The patient opted for surgery and so a DMSA scan was arranged in order to assess the split function of the kidneys. The left horseshoe kidney showed no function; RALP was planned with intraoperative ligation of the left distal ureter.\nAs per BAUS guidelines, during the COVID-19 pandemic, the patient was started on hormone treatment for prostate cancer for nearly 100 days whilst waiting to have surgery.\nAt the time of robotic prostatectomy, the surgeon adopted the posterior approach in order to identify the structures at the bladder base more easily. Both seminal vesicles were identified and dissected. The vasa were also revealed and cut. The left ureter was encountered near its opening into the left SV (). This was cross-clamped and cut. The operation was then completed using the anterior approach with right side wide excision and left intrafascial nerve sparing. Console time was 2:30 hours and no complications occurred. Estimated blood loss was 100 mls.\nPostoperatively, the patient's condition was stable and urinary output was good. Estimated GFR (kidney function) remained normal. The patient was deemed medically fit for discharge the following morning. Postoperative histology revealed pT3bNxMxR0 Gleason 3 + 4 prostate adenocarcinoma and confirmed the histology of the cut ureter (). The patient was reviewed 4 weeks postoperatively and had no loin pain. Follow up CT urogram showed no hydronephrosis in the atrophic left horseshoe kidney whereas there was normal uptake and excretion by the right kidneys. He was fully continent at 8 weeks with undetectable PSA. |
A 33-year-old, white, gravida 2, para 2 woman was referred to our Gynecology Department with intermittent abdominal pain that had intensified during the previous 3 months and progressive abdominal swelling during the previous 2 years. Her past medical and gynecologic history was otherwise unremarkable. No familial history of any disease was reported. A physical examination revealed a firm giant palpable abdominal mass with identifiable borders. The mass extended to her umbilicus and measured 15 cm above her symphysis pubis. These findings were confirmed by abdominal sonography. The abdominal ultrasonic examination revealed a markedly enlarged and lobular uterus containing intramural uterine leiomyomas, the largest measuring 17×15 cm without ascites in her abdominal cavity. No additional pathology was noted in the remainder of her pelvis or abdomen. The results of the routine laboratory testing, including a complete blood count, serum electrolyte levels and biochemical tests, were within normal limits. On the basis of these findings, a giant intramural myoma was assumed, and myomectomy was selected as the treatment.\nShe was offered laparoscopic removal of the myoma. Pneumoperitoneum was achieved using a supraumbilical Veress needle until an intra-abdominal pressure of 12 mmHg was reached. We first placed a midline supraumbilical 10 mm port for the telescope, and then two 5 mm accessory trocars were positioned in the left and right lateral quadrants visualized via a 10 mm telescope inserted through the supraumbilical port. The left and right accessory ports were inserted lateral to her deep inferior epigastric arteries and higher than usual; the accessory trocars were inserted sufficiently high enough to provide an unobstructed passage to the myomas for the laparoscopic instruments. Intra-abdominal visualization revealed an enlarged, lobular uterus containing a fundal intramural myoma with a maximum diameter of 17 cm, as diagnosed by ultrasound (Fig. ). The adnexa on both sides, round ligaments, and other pelvic and abdominal organs were normal. A myomectomy was performed using the standard technique as described elsewhere []. A vertical incision was made on the prominent part of the principal myoma using a monopolar hook. The cleavage plane between the myoma and its surrounding connective tissues was then dissected. When the myoma was identified, the myoma was fixed, and enucleation was then accomplished by traction on the myoma with a tenaculum clamp, associated with countertraction on the uterus-facilitated dissection. The myoma was completely enucleated and removed without disturbing the endometrial cavity. A Harmonic ultrasonic scalpel (Ethicon Endo-Surgery Inc, Cincinnati, OH, USA) was used for most of the procedure. Bipolar coagulation was used when extra hemostasis was required. The myometrial defect and edges were closed with a continuous suture using a V-loc unidirectional barbed suture (Covidien, UK) in two layers (Fig.). The left accessory 5 mm port was converted to a 10 mm one for the insertion of the morcellator. The entire myoma was removed using an electromechanical Rotocut G1 tissue morcellator (Karl Storz, Tuttlingen, Germany). The total intraoperative blood loss was 720 mL, the total weight of the myoma removed was 2005 g and the operation lasted for 140 minutes.\nThere were no major intraoperative complications. The final histopathological examination confirmed the diagnosis of a uterine leiomyoma. The postoperative course was unremarkable, and the patient was discharged on the second postoperative day. |
A 10-year-old boy was referred to the paediatric dental clinic with the chief complaint of the unpleasant appearance of a large anterior tooth. His medical history was noncontributory. There was no family history of dental anomalies, and there was no previous trauma to the teeth or jaws. The patient was a single child of a nonconsanguineous marriage.\nIntraoral examination revealed a large maxillary right central incisor, with a small groove observed on the labial and palatal aspects of the crown (). Response to pulp testing was normal, no caries were detected, and there was no significant periodontal pocketing. The patient was in mixed dentition and had crowding in the anterior region due to a lack of space. The total number of teeth was normal.\nRadiographic examination revealed fusion of the maxillary right permanent central incisor with a supernumerary tooth. According to the radiographs, the fused tooth had two distinct roots, with no connection between the pulp chambers and canals. No periapical radiolucency associated with these teeth was detected (Figures and ).\nAn orthodontic examination was conducted, and an initial treatment plan was developed that aimed to separate the fused supernumerary tooth and then correct the arch discrepancy with orthodontic treatment. The treatment objectives and alternatives were explained to the patient and his parents who provided their written consent prior to treatment.\nThe teeth were anaesthetized (Ultracain D-S, Istanbul, Turkey), and buccal and palatal flaps were raised. Some bones were removed below the buccal groove in order to locate the point at which the two roots separated. The crown was sectioned with a diamond bur (Midwest, Dentsply, IL, USA), and the mesial part of the tooth was removed (Figures and ). The root surfaces were examined, and no pulpal association was detected (). The remaining distal tooth was restored with composite resin (Gradia Direct, GC Corporation, Tokyo, Japan), and the flaps were sutured (). Following surgery, the patient was prescribed chlorhexidine mouthwash (Orasept, Biofarma İlaç San. ve Tic. A.Ş, İstanbul, Turkey). Sutures were removed one week postoperatively.\nFixed orthodontic treatment was then agreed in order to correct the position of the central tooth, and routine orthodontic records (plaster model, photographs, and radiographs) were obtained. Clinical examination and analysis of the records showed the maxillary right central tooth to be infrapositioned. Cephalometric analysis showed the upper and lower jaws to be in normal position in the sagittal plane in relation to the base of the cranium, but the right upper central tooth was found to be protruding slightly due to the fusion. In order to eliminate the malocclusion, a fixed orthodontic appliance was planned using 0.022 inch Gemini MBT metal brackets (3 M Unitek, USA). Following placement of the maxillary incisor brackets, an arch wire (16 Heat Active Nickel-Titanium, 3 M Unitek) was connected to the bracket slots to correct the position of the maxillary right central tooth ().\nAt the fixed appliance adaptation appointment, the patient was instructed on oral hygiene and prohibited foods and was called back for routine orthodontic controls once a month. When the maxillary right central incisor had moved into the correct position in the arch, 0.019′′ × 0.025′′ Heat Active Nickel-Titanium and 0.019′′ × 0.025′′ stainless steel arch wires were attached to correct the midline diastema. At the end of a 13-month followup period, orthodontic treatment using fixed mechanics was found to have achieved adequate positioning of the central teeth. Following fixed orthodontic treatment, a Hawley appliance was constructed for retention ().\nOnce orthodontic treatment had been completed, the maxillary central teeth were reshaped with composite resin (Gradia Direct, GC Corporation, Tokyo, Japan) for aesthetic reasons. Teeth were checked for possible complications once every three months. After an 18-month followup period, the maxillary right central tooth showed no sign of periapical pathosis (Figures and ) and responded positively to electric pulp testing. Followup examinations indicated that treatment had successfully restored both esthetics and function. Probing revealed no periodontal pocketing around the central incisor, and there was good attachment; however, long-term followup is required. |
The patient, a 28-year-old woman, was admitted to the Department of General Surgery, Sir Run Run Show Hospital, for half a month of splenic mass found by physical examination. Physical examination showed clear, spiritual, nonyellow skin and sclera, no swelling of superficial lymph nodes, no difference between cardiopulmonary examination, abdominal soft, no obvious mass, normal bowel sounds, no tenderness and rebound pain in the whole abdomen, negative mobile voice, negative Murphy's sign, negativity of renal percussion pain, no edema in both lower extremities, and negative pathological signs. In the imaging diagnosis of abdominal CT enhancement, the cystic space-occupying lesion of the spleen is considered (). We should improve the preoperative examination and exclude operative contraindications. During the operation, there was about 8 cm of the upper pole of the spleen, and the boundary was clear. . There was no obvious abnormality in the exploration of the abdominal viscera. The operation was successful. The operative time was 115 minutes, and the blood loss was 20 ml. On the first day after the operation, the patient took a liquid diet. The patient had anal exhaust in the second day and the patient was discharged four days later. Postoperative pathology revealed an epidermoid cyst.\nThe patient was placed in an incomplete right lateral decubitus position. A pneumoperitoneum of 12 mmHg was established using a Veress needle through a left paraumbilical incision, and an optical port (5- to 12-mm trocar) was introduced afterward. Under visual control, two robotic 8 mm trocars were placed in the left hypochondriac region and the epigastrium. An additional 5- to 12-mm accessory port was placed in the left lumbar region on the middle axillary line for the side assistant surgeon. The surgical cart with the robotic arms was positioned on the patient's left side at a 45° angle to the table's longitudinal axis.\nAs reported by the European Association for Endoscopic Surgery clinical practice guidelines, in preoperative imaging, splenomegaly was defined with a maximum splenic diameter of more than 15 cm []. Postoperative morbidity has been specified as any complication that occurs within 30 days of surgery and has been evaluated as in [].\nDissection was performed with the robotic EndoWrist® Fenestrated Maryland Bipolar Cautery on the left hand and HarmonicTM Curved Shears on the right hand. The trocar position is presented in . The table side assistant elevated and moved the spleen or provided suction if needed. In one case with the large hydatid cyst, it was first inactivated and evacuated under visual control in order to increase the working space in the upper abdomen. After dividing the peritoneal attachments and the splenic ligaments with the HarmonicTM Shears, the omental bursa was opened and the splenic vessels were dissected in the hilum. In the three cases of subtotal splenectomy, the splenic artery and vein were ligated with intracorporeal knot tying using EndoWrist® Needle Holders. After complete mobilization, the splenic parenchyma was transacted using an Endo GIA Roticulator™ blue cartridge stapler introduced by the side assistant surgeon through the accessory port. Vascularization of the splenic remnant was based on the anastomotic branch of the left gastroepiploic pedicle.\nThe specimen was removed in an Endo Catch™ II 15 mm specimen pouch through the accessory port incision. Hemostasis on the transection surface of the splenic remnant was completed in two cases with a TachoSil® hemostatic sponge.\nThe drain tube was removed after 1 day. The patient was discharged on postoperative day 4 with no postoperative complication. And the final pathological findings showed epidermoid cyst. We performed a follow-up visit of 1 year, and no recurrence or abnormal platelet count was found.\nWith the popularization and application of ultrasound, abdominal CT and magnetic resonance imaging in clinical practice, and the awareness of public health examination, the number of splenic cysts diagnosed in recent years has increased significantly. According to the etiology, splenic cysts can be divided into two categories: nonparasitic cysts and parasitic cysts. In nonparasitic cysts, splenic pseudocysts and splenic true cysts can be classified according to the presence or absence of lining epithelium in the cyst wall. Pseudocysts are more common. Most of them are formed after trauma secondary to subcapsular hematoma of the spleen. They are also called secondary splenic cysts. The wall of the cyst is only a fibrous tissue without lining epithelium. The cyst contains blood or serous fluid. The splenic cyst is a rare disease with unknown etiology. The inner wall of the cyst has lining epithelium. The cyst can be unilocular or multilocular. According to pathology, it can be divided into four types: epidermoid cyst, dermoid cyst, cystic lymphangioma, and cystic hemangioma. Ultrasound examination is often the first choice for splenic cysts because of its nonradiation, low cost, and convenient examination. The images are mostly cystic masses with clear borders and dark areas without echoes. After the detection of splenic space-occupying lesions by ultrasonography, CT, MRI, and other imaging examinations can further understand the size, shape, and type of splenic cysts. The number, characteristics of cyst cavity and wall, and adjacent relationship with surrounding organs are of great value.\nSurgical treatment is the main method for a splenic cyst. Clinically, nonparasitic splenic cysts less than 2.0 cm in diameter and without any symptoms can be observed regularly. When the diameter of splenic cyst is more than 5.0 cm, the follow-up of the outpatient department increases rapidly or complications such as compression of surrounding organs, rupture, and infection occur; timely surgical treatment is advocated. At present, the main surgical methods depend on the size and location of the cyst, including fenestration and drainage of splenic cyst, total splenectomy, splenectomy with preservation of accessory spleen, hemisplenectomy, and partial splenectomy. As the main immune organ of the human body, the spleen plays the role of regulating immune function and clearing senile blood cells. If total splenectomy is performed, the chance of overwhelming postsplenectomy infection will increase, and complications such as platelet elevation and venous thrombosis will be more likely to occur. Therefore, it is advocated that surgery should preserve the function of the spleen as far as the patient is concerned, such as the presence of a cyst on the upper pole of the spleen or the presence of a cyst at the lower part of the spleen. The diameter of the spleen is less than 50% of the total volume of the spleen. The partial splenectomy or partial splenectomy can be performed according to the specific conditions of the patients. |
A 74-year-old woman visited our station with squamous cell carcinoma (SCC) on the right buccal mucosa. Her past medical history included chronic obstructive airways disease, hypertension, and diabetes mellitus. The patient is a current smoker, with a history of 20 pack-years. Preoperative chest radiography, electrocardiogram, full blood count, and serum biochemistry were within the normal range. After being diagnosed with SCC as a result of incisional biopsy, the patient underwent the resection of SCC on the right buccal mucosa of the mandible, modified radical neck dissection, and primary reconstruction with a fibula-free flap. Tourniquet pressure was 300 mm/Hg, and its application time was 60 min. Total on-table time was approximately 7 h. Upon admission to the SICU after the 7-h operation, hypothermia and hypotension were noted. On the first postoperative day, the patient exhibited oliguria and proteinuria and elevation of CK, AST, ALT, and LDH. Together with the nephrology and neurology staff, we tried to figure out our patient’s symptoms and clinical findings. We thought that her clinical picture was based on an impression in which acute renal failure was diagnosed as secondary to rhabdomyolysis. Thus, she was managed with high-dose loop diuretic therapy. Additionally, we gave her hepatotonic to recover her liver function. The patient was supplemented with 150 to 250 mL/h of lactated Ringer’s solution and 0.9% NaCl. When the volume was full, urine output of above 100 mL/h was maintained by 20 mg intravenous injection with furosemide.\nHer urine output for the first hour is at 20 mL/hour, but after the medication, her urine output began to improve on day 4 with a corresponding reversal in the serum creatinine. After postoperative day 4, the muscular enzyme showed a downward trend. We treated the patient with medication and hydration, and then the result became favorable. In the end, she was able to recover fully from the symptoms. Figures and show the change in serum enzyme levels during hospitalization (Figs. and ). |
A 78-year-old man presented to our emergency department because of acute onset chest pain that started two hours prior to presentation. The pain was retrosternal, pressure-like, moderate in intensity and started upon awakening from sleep. His past medical history is significant for type II diabetes mellitus and pancreatic cancer that was treated with the Whipple procedure 27 years ago. He had coronary artery disease status post percutaneous angioplasty with stenting of the mid right coronary artery (RCA) 12 years ago, and stenting of the proximal circumflex and proximal RCA 17 years ago. A paclitaxel drug-eluting stent (PES) 3.0 x 24 mm was used to stent the mid RCA 12 years ago (Figure ).\nThe patient is a former cigarette smoker, and does not consume alcohol, caffeine, or illicit drugs. At presentation, he was in mild distress, complaining of typical chest pain persistent despite aspirin administration. On physical examination, the patient was noted to be diaphoretic. His heart rate was 60 bpm, and blood pressure was 110/75 mmHg. Electrocardiogram (ECG) showed ST-segment elevation in the inferior leads II, II, and avF (Figure ).\nThe patient was loaded with aspirin and clopidogrel and emergently taken to the cardiac catheterization laboratory. Coronary angiography showed thrombotic occlusion of mid RCA DES placed 12 years ago. Immediate percutaneous coronary balloon angioplasty was performed followed by a 3.5 x 16 mm everolimus drug-eluting stent (EES) deployment at a maximum inflation pressure of 14 atm. Following the intervention, excellent angiographic appearance of the artery was obtained with a 0% residual stenosis (Figure ).\nDue to the acuity of the situation and the patient’s unstable hemodynamic status, it was difficult to obtain intravascular images of the lesion and tell with certainty what mechanism led to this event. The patient was stabilized and monitored for the following 24 hours with no further complications, and was then successfully discharged home. |
A 55-year-old Caucasian male, with a past medical history significant for tobacco abuse (41 pack-years), presented with shortness of breath accompanied by chest and back pain for two months. Blood workup showed a WBC count of 68,400 cells/µL, with an AEC of 27,360 cells/µL. A computed tomography (CT) pulmonary angiogram was performed, as he was hypoxic, and revealed a 3.6-cm speculated mass within the anterior right upper lobe, partially invading the anterior chest wall. It also revealed mediastinal and hilar adenopathy, an extensive osseous lesion (including compression fracture at T7), and a small pericardial effusion (Figure ). A CT of the abdomen and pelvis with contrast was performed and revealed a diffuse metastatic disease involving the liver, adrenal glands, spleen, and the bones. Magnetic resonance imaging (MRI) of the thoracic spine did not reveal spinal cord compression, but it did show the compression fracture at T7 and multilevel thoracic spondylosis. An MRI of the brain revealed a 5-mm lesion in the left occipital lobe, without edema or mass effect.\nThe hematology-oncology team was consulted for an evaluation of the metastatic disease and the eosinophilia. A core needle biopsy was obtained from a liver lesion and the result came back as poorly differentiated adenocarcinoma of the lung (cytokeratin 7, TTF1, and napsin-A were positive, while cytokeratin 2 and CDX2 were negative). Given his functional status, the decision was made to hold on systemic therapy and start on palliative radiation to the spine for pain control. The plan was to complete radiation sessions and then evaluate his functional status before starting systemic therapy.\nHe continued to have a high WBC count during the admission (Figure ). Therefore, a bone marrow biopsy was performed to rule out a hematologic malignancy and it revealed metastatic adenocarcinoma of the lung with no evidence of a myeloproliferative disorder. The flow cytometry from the bone marrow showed a CD5-positive clonal B-cell population, which was similar to the blood flow cytometry. Blood tests, including tests for Janus kinase 2 (JAK-2), calreticulin (CALR), MPL, BCR-ABL, and platelet-derived growth factor receptor (PDGFRA), were negative. The blood smear showed microcytic anemia with leukocytosis with absolute neutrophilia and eosinophilia. The serum immunoglobin E (IgE) was high at 377 IU/ml, and the tryptase level was low at 1.8 µg/L. Given these findings, his eosinophilia was related to a paraneoplastic process rather than a primary bone marrow disease.\nDuring the following days, the patient completed 13 sessions of radiation without improvement in his functional status, pain, or breathing. The case was discussed with the patient and his family; he decided that he would go with comfort measures, so he was discharged to the hospice facility. |
On April 30, 2014, a 46-year-old male patient was admitted to the gastroenterology department with dyspepsia, abdominal pain, vomiting, and a 4 kg weight loss. The patient had previously undergone an appendectomy and abdominal abscess debridement for right abdominal pain and fever in January 2008. Seven months after the operation, he underwent a resection of the ileocecum for a spontaneous perforation that occurred in August 2008. The pathological diagnosis was ITB and the patient underwent antituberculosis therapy for 1 year.\nThe patient was hospitalized for right lower quarter pain on April 30, 2014. He had a fever, and physical examination revealed a bulge on the right lower abdomen, obvious tenderness, no fluctuation, no rebound pain, and muscle tonus. The laboratory results for the patient revealed the following: a white blood cell count of 11.77 × 109/L (normal range 4–11 × 109/L), thrombocytosis (platelet concentration of 469 × 109/L, normal range 100–300 × 109/L), and a fecal occult blood test of 1+. All other tests were normal. The computer tomography (CT) scans showed a segmental and irregular thickening of the anastomosis, ascending colon, and hepatic flexure with thickened mesenteric fat; dense shadows of anastomosis; and multiple stripes, nodes, exudation, and bubbles. Colonoscopy revealed a severe hyperemia and edema of mucosa in the anastomosis that was associated with inflammation, a fistulous opening, mucopurulent exudate, and multiple deep ulcers. The pathology showed mucosal chronic inflammation associated with ulcers and acute inflammation, moderate atypical hyperplasia of the glands, and a crypt abscess. The patient became asymptomatic after anti-inflammatory and nutritional support therapy.\nThe patient was hospitalized for right lower quarter pain again on June 15, 2014. His vital signs were normal and physical examination revealed a mass on the right lower abdomen (5 × 4 cm in size). All laboratory tests were normal. The CT scan showed the wall thickness of the right colon and the anastomosis, and the gas shadow in swelling soft tissue of the right lower abdominal wall (Figure ). A right hemicolectomy was performed after the preparation because of the persistent right lower abdominal mass. Macroscopic histopathology showed that the intestinal wall was thickened with luminal narrowing (Figure ).\nThe pathology after the operation showed colic mucosal ulceration along with transmural inflammation, mixed inflammatory infiltrate, and granuloma (ie, lymph cells and plasmacytes infiltrated the abdominal wall, and a granulomatous change occurred in the muscular layer) (Figure ). The Ziehl–Neelson stain for acid-fast bacilli was negative. As the time between the first diagnosis and the second diagnosis was 6 years, 2 of our pathology experts rechecked the pathological data with the permission of the patient and the hospital. Both experts considered that the pathological diagnosis should be CD rather than ITB. Ultimately, we made a diagnosis of CD, which is indicative of the pathological changes and the history of recurrent intestinal fistula. According to the patient's CDAI, he was treated with infliximab. At the time of the 8-month follow-up, the patient had remained in complete remission and made a good recovery. |
The patient is a 51-year-old Turkish man who recently presented to our institution with acute left flank pain and hematuria. Past history is significant for an atrophic nonfunctioning right kidney and a functioning left kidney with longstanding ureteropelvic junction stenosis. Since 1992, the patient has undergone an open dismembered pyeloplasty, 2 endopyelotomy attempts, and several stone treatments with ureteroscopy, holmium laser, and basket extraction.\nDuring the second endopyelotomy attempt in 1992, a left renal subsegmental arterial branch was inadvertently traversed, resulting in acute hemorrhage and development of an arterial-ureteral fistula. This was embolized by interventional radiology with bare metal coils, with satisfactory hemostasis. A left ureteral stent was subsequently placed for the chronic ureteral obstruction.\nAfter this procedure, the patient underwent routine ureteral stent changes every 2 years, which is a much longer period than that during which stents can typically be left in place before problems such as encrustation, infection, or obstruction are estimated to develop (2–4 months). Indwelling 7/14 French 28-cm endopyelotomy stents are used.\nIn 2010, the patient presented to our institution's emergency department (ED) with significant hematuria and flank pain. Computed tomography (CT) of the abdomen and pelvis without intravenous contrast (stone protocol) was performed and showed several small oval and tubular high densities in the left renal pelvis, all of which were initially interpreted as renal calculi (). The preexisting ureteral stent appeared intact and stable since the prior CT. The patient was then brought into the operating room for stent change and stone retrieval.\nIntraoperatively, at least 2 bare metal coils were observed to endoscopically erode into the left renal pelvis. Few small renal calculi were also seen, most likely due to the coils acting as the nidus. Upon further review of the CT with multiple different windowing levels, the coils could be subtly distinguished from the stones owing to their tubular appearance and configuration of the tightly wrapped loops. |
A 71-year-old Asian male lung cancer patient, with total obstruction of the right upper bronchus, underwent right upper sleeve lobectomy with bronchoplasty and lymph node dissection combined with preoperative induction and postoperative adjuvant chemotherapy with docetaxel and platinum agents in 2007. Thereafter, partial resection with right radical neck dissection was performed for tongue cancer of pT1N1M0. In 2016, a new undiagnosed hilar mass near the anastomosis site, as a lung metastasis of tongue cancer found on chest computed tomography (CT), was treated with radiotherapy of 36 Gy at a previous hospital. The tumor responded transiently to radiotherapy, but progressed. On chest CT at the first visit in 2018, a tumor involving the remaining two lobes compressed the pulmonary artery at the cut end of the superior trunk (Fig. a, b). Moreover, the tumor extended from the periphery of the anastomosis site to the tracheal bifurcation and to the inflow to the superior vena cava (SVC) of the azygos vein (ligated previously), which was ligated previously, along with the membranous side of the right main bronchus (Fig. c, d). In 2016, chest CT on initial detection of recurrence suggested lobar lymph node recurrence near the anastomotic site. On bronchoscopy, the bronchial anastomosis site was on the peripheral two rings from the tracheal bifurcation; tumor infiltration was suspected in the immediate periphery on that membranous side, but histological diagnosis could not be obtained from biopsy on this site. Distant metastasis was not observed in fluorodeoxyglucose-positron emission tomography, and the clinical stage was stage IB of cT2aN0M0, if it was a primary lung cancer. The high-risk patient had no abnormality in the electrocardiogram and the echocardiography in spite of having a history of coronary vasospastic angina; however, both %ppo-FEV1 and %ppo-DLco were slightly less than 40% (38.0 and 37.8%, respectively) in the pulmonary function test. Because radiotherapy has already been performed by the previous hospital and appropriate drugs could not be selected as there was no information on definitive diagnosis and gene mutation, we selected the most effective surgical treatment despite being a high-risk patient. For surgical management, after securing and cutting the proximal side of the main pulmonary artery with a median sternotomy, the tumor in contact with the azygos vein stump was divided safely from the SVC, and sleeve pneumonectomy was performed under good vision via the posterior lateral thoracotomy approach.\nMedian sternotomy in the supine position was performed under general anesthesia. The proximal part of the right main pulmonary artery could only be secured in front of the left main bronchus because of severe adhesion from the tracheal bifurcation to the right main bronchus. The right main pulmonary artery was occluded for approximately 15 min to prevent deterioration of circulatory dynamics. Although the remaining lymph nodes around the trachea were dissected and the SVC was adequately detached (Fig. a), confirming the adhesion between the tumor and azygos vein was difficult under direct vision from this position. After dividing the proximal site of the right main pulmonary artery with a vascular stapler (Fig. b), posterior lateral thoracotomy was performed in the left lateral position. The azygos vein stump, which was in contact with the tumor, could be observed from the thoracic cavity side. After the inferior pulmonary vein and middle lobe vein were cut, the area around the stump of the azygos vein was peeled off and divided at the edge of the bifurcation from the SVC using vascular stapler (Fig. c). As the bronchial stump proximally from the anastomosis showed infiltration of cancer cells by frozen section, sleeve pneumonectomy was performed. The left main bronchus and distal trachea were exposed and mobilized with blunt dissection to avoid excessive peeling and preserve maximal blood supply. After dividing the left main bronchus, a spiral tube was intubated from the operative field; the tracheobronchial sleeve above the carina was then resected (Fig. d). The distal and proximal margins were confirmed radical by frozen sections. Reconstruction was performed by telescoping the left main bronchus into the distal trachea to overcome marked caliber mismatch. The distal trachea, around the anastomosis, has thickening and low-mobility area, so the first three sutures at the deepest anastomosis edge were knotted extraluminally and 15 interrupted stitches with a 3–0 PDS were placed alternately from both sides (Fig. e). After tying the sutures, the anastomosis site was covered with pericardial fat pads.\nPostoperative pathological examination diagnosed a recurrence of lung squamous cell carcinoma because of its similarity to the previous histologic type. A tumor measuring 40 × 32 × 30 mm in diameter had central necrosis because of radiotherapy. The resected lymph nodes were free from metastasis.\nPostoperatively, treatment for circulatory insufficiency associated with arrhythmia and pneumonia was required (Fig. f), but the patient was transferred to a public hospital on postoperative day 87 for rehabilitation. He is still alive during the 13 months after the operation without anastomotic problems and recurrence. |
A 19-year-old male patient who was a victim of a robbery and was hit by a gunshot with an entry wound behind his ear presented with a right comminuted condylar base process and zygomatic arch fractures []. This study was approved by the Local Research Ethics Committee under approval number 524680, and all participants signed informed consent agreement. The patient who appears on the images has signed informed patient consent authorizing the publication of his images.\nAll patients required immediate treatment to prevent site infection and were treated with the same surgical procedure [Figure and ] that consisted of an extended preauricular incision, removal of the proximal and fractured small bone fragments (including the condylar head), removal of any foreign body, reshape/flattening of the fractured surface of the distal fragment, fixation of the remaining disc (if viable) with a nylon 2-0 sutures anchored to two 2.0 mm screws (inserted posteriorly and laterally to the condyle neck), lining of the TMJ with temporalis fascia, application of Erich arch bars, and early elastic therapy. shows a step-by-step description of the surgical technique.\nAll patients showed a similar outcome: a good mandibular motion (>35 mm), satisfactory occlusion with teeth in maximum intercuspation, deviation to the affected side during mouth opening, slightly roll rotation of the occlusal plane, painless, and no infection.\nReduction in the mandibular ramus height was compensated by muscular and dental adaptations.[] Maximum intercuspation resulted from dental extrusion on the unaffected side and dental intrusion on the affected fracture side, causing a small rotation of the occlusal plane. Despite that, dental adaptations were mild and did not cause any periodontal or noticeable esthetic problem. Elastic therapy was monitored twice a week and was started with 2–3 Class II elastic bands, gradually removed until patients could keep a stable and reproducible occlusion, which lasted a maximum of 4 weeks. Patients were followed-up for a period of 6–12 months, and their occlusion and mouth opening were monitored. None of them showed any important reduction in mouth opening or premature occlusal contact/open bite as shown that describes the findings of Case 1. |
A 19-year-old female patient presented with a chief complaint of pain in left lower back tooth region since 10 days. On intraoral examination, mandibular left first premolar showed a large Class I caries involving pulp. The tooth was tender on percussion and showed no response to electrical and thermal pulp testing. Radiographic evaluation with intraoral periapical (IOPA) radiograph showed periapical radiolucency in relation to lower left first premolar []. More than one root canal was suspected in that tooth and extra medially and distally angulated periapical radiograph was taken to help in a better interpretation of the canal system. Based on the clinical and radiographic evidences, it was diagnosed as chronic apical periodontitis in relation to the mandibular left first premolar.\nAnesthesia was achieved by means of inferior alveolar nerve block with 1.8 ml of 2% lignocaine with 1:80,000 adrenaline. Teeth were isolated using rubber dam and all caries were removed prior to access opening. After access opening, the trunk of the canal seemed to trifurcate at mid-root level giving rise to three separate canals. The orifices to the three root canals were found and enlarged with Gates Glidden Drill. The pulpal tissue was removed using barbed broach; the working length was determined using periapical radiograph. Working length radiograph revealed three canal orifices leading to three separate canals but exited as two apical foramina's []. The three root canals were thoroughly instrumented and shaped by step back technique to a size 30 (apical preparation), and the root canals were irrigated using 3% sodium hypochlorite and 3% hydrogen peroxide solutions during root canal cleaning. The teeth were then dried with sterilized paper points. Intracanal medication with calcium hydroxide powder mixed with saline, carried through lentulo spirals, was placed. At the second appointment, the root canals were obturated with zinc oxide sealer and laterally condensed Gutta-percha []. A final radiograph was then taken to confirm the quality of the obturation []. The access cavity was sealed with IRM and the patient was then given appointment for permanent restoration. |
A 42-year-old white man had a history of kidney cancer from 2005. At that time, his right kidney was removed, and RCC was concluded on the basis of histology. The disease was staged as T3apN0M0 (TNM-Union for International Cancer Control, TNM-UICC, 2010). The cancer was regarded as localized and no further investigation was undertaken at the time. In the winter of 2013, the patient consulted his general practitioner for increasing right-sided lateral chest pain. Computed tomography (CT) of his thorax revealed a lateral mass destructing his third rib, without penetrating through the pleural cavity. Several biopsies were obtained and the tumor was confirmed as clear cell RCC. However, the pathologist responsible had doubts about the correct histology and consulted experts in the field for a second opinion abroad. Techniques including Sanger sequencing of the candidate translocation region and fluorescence in situ hybridization (FISH) confirmed a translocation including TFEB (t6;11), which is diagnostic of RCC. Immunohistochemistry was positive for cathepsin K and Melan-A, which supported the diagnosis.\nFurther investigation of the patient with magnetic resonance imaging (MRI; Fig. ) of his vertebral column showed small lesions in his lower lumbar spine, suspicious of tumor spreading to his skeleton. This clinical picture, and the first immunohistochemical results showing intensive staining of cathepsin K, pointed to dysfunctional activity in osteoclasts, and linked therapeutic choices to autophagy and metabolism. He was referred to surgical resection of the part of his chest wall afflicted by a tumor mass, which was conducted at the University Hospital Oslo in September 2013. Postoperatively, he received irradiation to the tumor bed, 3 Gy per fraction to a total dose of 30 Gy.\nToday, there is no commonly accepted treatment recommendation for non-RCC. Pending the final histology report, while having the clinicopathological suspicion of translocational RCC, several therapeutical strategies were discussed. In the light of no available standard treatment for patients with translocational RCC, a mammalian target of rapamycin (mTOR) inhibitor (everolimus 10 mg daily) counteracting phosphorylation of TFEB by mTOR complex 1 (mTORC1) [] and denosumab (120 mg subcutaneously every fourth week), a monoclonal antibody regulating osteoclast activity by targeting the RANK ligand, were both applied during and after postsurgical radiotherapy. This therapeutic strategy aimed to counteract the dysfunctional signaling effect in catabolism and bone reabsorption as described for TFEB tumors. In addition, the formerly nephrectomized patient had moderately reduced kidney function after the operation, and needed self-catheterization. Often tyrosine kinase inhibitors (TKIs) affect kidney function, supporting an mTOR pharmacological treatment instead of TKI for this patient. Unfortunately, everolimus led to thrombocytopenia and had to be reduced to 7.5 mg per day taken orally. During the next 2 months, the patient’s health deteriorated, and the mTOR-inhibitor therapy was eventually interrupted.\nA new series of MRI (Fig. ) confirmed progression, with multiple additional bony lesions. The patient was treated with a broadly acting TKI, pazopanib (800 mg once daily) as second-line medical treatment, interfering with platelet-derived growth factor (PDGF) and vascular endothelial growth factor (VEGF) pathways. At the same time, neither anti-1-amino-3-18F-fluorocyclobutane-1-carboxylic acid (18F-FACBC) positron emission tomography (PET)/CT nor 2-deoxy-2-(18F)fluoro-D-glucose (18F-FDG) PET/CT scans detected tumor spreading.\nAcknowledging the difficulty in identifying a rational therapeutic approach, the patient consented to serial biopsies taken in February and June 2014, preceding the choice of a new treatment due to a major lesion in his sacrum. Both samples were then assessed molecularly by Agilent SureSelect exome capture with Illumina sequencing, and expression profiling using bead arrays from Illumina.\nRibonucleic acid (RNA) extraction from the tumor taken at the first time point was split in two parts and a TissueLyser (Qiagen) was used to disrupt the sample. RNA from both parts was extracted using a GenElute Mammalian Total RNA Miniprep Kit (Sigma-Aldrich), according to the manufacturer’s instructions. DNA was extracted from the tumor taken at the second time point, using the NucleoSpin Tissue Kit (Macherey-Nagel), according to the manufacturer’s protocol. Normal DNA was extracted from ethylenediaminetetraacetic acid (EDTA) blood using the NucleoSpin Blood Midi Kit (Macherey-Nagel), according to the manufacturer’s protocol. Normal and tumor DNA was subjected to whole exome sequencing using the SureSelect whole exome v5 and Illumina sequencing by synthesis technology (HiSeq 2500) following the supplier’s protocol. The resulting FASTQ files were further analyzed using an in-house developed pipeline for somatic event detection. Reads of the tumor and its matched control sample were separately mapped with BWA-MEM [] to human reference genome (build b37) with an added decoy contig, obtained from []. Sample-wise sorting and duplicate marking was performed on the initial alignments with Picard tools []. Genome Analysis Toolkit (GATK) tools [] were subsequently used for two-step local realignment around indels (in this step, both samples were processed together). Each sample’s pair-end read information was then checked for inconsistencies with Picard, and base-quality recalibration was performed by GATK.\nSomatic variant calling on the sample pair was done with MuTect [] (somatic single nucleotide variant, SNV, detection), Strelka [] (somatic SNV and INDEL detection), DELLY [] (large-scale variation – deletions, duplications, translocations and inversions – detection) and VarScan2 [] (somatic copy number variation, CNV, analysis).\nGATK tools were used for computing coverage statistics based on the recalibrated alignment files.\nMost of the analysis (starting with the local realignment step) was limited to exome regions, defined by Agilent sequencing probes (for further details, see Additional file ).\nOne translocation was identified through sequencing, involving positions chr11:65,267,772 and chr6:41,659,234, involving the expected TFEB translocation previously described for this patient.\nWe identified 23 somatic single SNVs, and three insertion/deletion events. Among these, one mutated gene was located in the tumor necrosis factor (TNF) receptor pathway (MAP3K7, G110V), a pathway previously implicated in clear cell RCC []. However, no obvious candidate therapeutic target genes were identified as mutated.\nA DNA copy number plot is shown in Fig. . Gain of a region of 1q including the gene KIF14 has previously been associated with fatal progression, and KIF14 was among the genes reported to be overexpressed in tumors with 1q []. We did identify a somatic mutation for this gene (H849Y), but the tumorigenic potential remains unclear. Other chromosome arm scale events with more or less clear breaks could be seen on chromosomes 1, 3, 18 and 22. Some chromosomes (9, 13, 19) appeared to be generally underrepresented by sequencing reads in the tumor, while some other chromosomes (for example 2 and 7) seemed to be affected by many smaller events.\nTranscriptome analysis was performed using the TruSeq Stranded Total RNA with Ribo-Zero Gold from Illumina according to the manufacturer’s instructions. The RNAa-Seq library was sequenced using a HiSeq 2500 with a paired-end 2×100 base pair (bp) approach. The sequencing reads were mapped with TopHat2 [] to human reference genome/transcriptome (build hg19) and subsequently processed by the Cufflinks2 tools [] (cufflinks, cuffmerge, cuffdiff) in order to generate a gene-wise fragments per kilobase of transcript per million mapped reads (FPKM) expression (Table ).\nA plot of normalized transcript counts is shown in Fig. . In order to estimate upregulation of transcription compared to a normal kidney, we obtained transcript counts from the RNA-Seq Atlas [] (Fig. ). In order to evaluate whether there was an expression effect of TFEB being overexpressed, we examined whether genes that previously had been reported as upregulated in transiently TFEB-overexpressed HeLa cells were also affected in the tumor (Fig. , TFEB curve). A marked curve shift towards higher transcript levels, compared to the full list of genes, was observed, indicating that the high TFEB level observed in the tumor also increased expression of the reported target genes. We also examined whether a previously reported gene set for coordinated lysosomal expression and regulation (CLEAR) was upregulated in the tumor, and this could also be observed. Moreover, TFEB expression is very high in the tumor, compared to TFEB expression levels observed otherwise in normal kidney tissues [, ].\nThe transcriptional profile confirmed the clinical suspicion of highly upregulated macro-autophagy and dysfunctional pathway activity in c-MET, MAPK, TSC2 and S100A9, and downregulation of mTOR, as previously reported relevant for this type of tumor (Fig. ). Treatment with an autophagy-inhibiting agent, hydrochloroquine 200 mg twice a day, was therefore started and subsequently increased to 400 mg twice a day, but stopped when progression was observed in July 2014.\nUnfortunately, 5 months after the start of pazopanib, the CT and MRI evaluation confirmed progression, and the treatment was switched to another broad-acting TKI, sunitinib dose 37.5 mg per day, as third-line therapy.\nIn the wake of the genetic results and the approval of checkpoint inhibitor therapy, a second course of palliative radiotherapy was planned in late July 2014 (see Fig. ).\nThe second biopsy results gained from DNA pointed to enhanced autophagy gene signature and MAPK and AKT pathways downstream. On histological examination, cathepsin K and Melan-A were highly positive. Due to local pain in the patient’s chest wall, a second course of three-dimensional CT conformal radiotherapy encompassing pleural metastases (3 Gy per fraction, 5 days per week) was planned 4 weeks later, and sunitinib treatment was halted prior to radiotherapy. Acute pleural bleeding confirmed with a CT scan showed tumor growth after 8 fractions of radiotherapy, and further irradiation was abolished. Sunitinib treatment was discontinued.\nIn view of the young age of the patient and the specific staining for Melan-A expressing tumor cells [] (Fig. ), he was finally accepted for ipilimumab, targeting CTLA-4, activating the immune system, combined with third-line sorafenib treatment, a kinase inhibitor used in the treatment of renal cancer. Ipilimumab was withdrawn rapidly, due to controversies around combined treatment in the physicians group, exaggerated by temporary health deterioration of the patient. At last follow-up, 16 months after the diagnosis of translocational RCC, the patient’s sorafenib treatment stopped due to disease progression and he died shortly afterwards.\nAttempts of cell culture of excess cell material from the bone biopsy in medium were unsuccessful. |
Mr. X, 20-year-old male patient, with pre-morbid anxious personality traits presented with an insidious onset illness of 1 year duration characterized by hearing music in his head. On exploration patients explained that, whenever he would attempt to concentrate in studies or try to do any activity requiring focused attention and concentration, he would have repeated intrusion of the melodies of recently heard songs. The songs/melodies would be of a particular singer with some sensual content in their lyrics or videos. The songs/lyrics/melodies ran in his mind against his wish, attempts to resist would evoke anxiety. This would occur repeatedly in the day for about 5-10 min at each occasion or shorter if he succeeded to distract; would end-up wasting about 1-2 h daily battling with this symptom. Over the period these symptoms kept on increasing and would occur anytime of the day and led to marked dysfunction and influenced his performance in the examinations. After 2-3 months of onset of these symptoms, he started remaining sad; his interaction with the family members reduced and preferred to sit alone in his room. His sleep was also disturbed with difficulty in initiation of sleep; appetite reduced and developed ideas of worthlessness. Over the period of next 2-3 months, with the onset of depressive symptoms, the intensity, frequency and duration of the hearing music increased. He once tried to end his life by strangulating himself but could not complete the act. He was seen by a psychiatrist after 1 year of onset of symptoms, was prescribed tablet desvenlafaxine 50 mg OD with which patient perceived 20-25% improvement in symptoms over the period of 8 weeks. Due to lack of further improvement, patient came to our hospital.\nOn the basis of available information a diagnosis of obsessive compulsive disorder and severe depression without psychotic symptoms was considered. He was managed with tablet escitalopram 10-15 mg/day. Additionally behavior therapy in the form thought stopping was done. Over the period of 6 weeks, patient achieved remission of his depressive symptoms. His musical obsessions also reduced significantly. However, 4 months later, due to pressure of examination his symptoms of musical obsessions worsened and resultantly dose of tablet escitalopram was increased to 30 mg/day and thought stopping was supervised. With the above over the period of 12 weeks he achieved remission. |
A 49-year-old woman with a past medical history of pulmonary embolism presented for IVC filter removal, which was initially placed approximately three months prior to this hospitalization. She was not a smoker and had no history of chronic lung disease or occupational hazards. At that time, the patient had right leg deep vein thrombosis and right sub-segmental pulmonary artery embolism. Hematological workup was inconclusive and it was presumed to be provoked by prolonged bed rest in the setting of bariatric surgery. The patient had a history of intracranial aneurysm in the posterior circulation so long-term anticoagulation was not thought to be a safe option. She got IVC filter placed via the right femoral approach.\nIn the present hospitalization, interventional radiology (IR) guided IVC filter removal was done in the operating room (OR). Under X-ray fluoroscopy, a snare was inserted into the internal jugular vein, and the IVC filter was withdrawn from its hook (Figure ). Per the OR notes, it was a difficult approach requiring manipulation of the snare. The patient was transferred after the procedure into an observation room.\nWithin an hour after the procedure, she developed pleuritic chest pain, hypotension, presyncope, and shortness of breath. She denied any other systemic symptoms like nausea, vomiting, diarrhea, joint pain, and bowel or bladder symptoms. Her physical examination showed a blood pressure of 85/60 mmHg, a pulse of 122 beats per minute, a respiratory rate of 26 breaths per minute, and an oxygen saturation of 92%. She had a poor inspiratory effort. Her jugular venous pressure was elevated (9 cm of H2O). On cardiovascular examination, her heart sounds were markedly diminished, and the point of maximal impulse was nonpalpable. No murmurs, gallops, or rubs were appreciated. Her chest was clear on auscultation. The results of her abdominal and neurological examinations were also unremarkable. Her pertinent laboratory findings revealed PT/INR of 3.4, a potassium level of 3.1 mEq/L, and hemoglobin of 10.6 g/dL. The other laboratory tests were unremarkable. Her chest X-ray revealed low lung volumes with bibasilar subsegmental atelectasis. An electrocardiogram (EKG) revealed sinus tachycardia, electrical alternans, low voltage QRS complexes, and a prolonged QT interval (Figure ).\nBedside echocardiography (echo) at the symptom onset showed a large anterior, small to moderate lateral and posterior pericardial effusion. Left ventricular and right ventricular functions were grossly normal. Transthoracic echo revealed moderate to severe pericardial effusion, 2.1 cm in the largest dimension with right ventricular collapse during diastole consistent with the tamponade effect (Figure ). It also showed a large echogenic mobile structure, consistent with pericardial thrombus.\nShe was started on intravenous fluid resuscitation and immediately transferred to the OR. With an emergent pericardiocentesis, 300 mL of blood and a large clot was removed, and a pericardial window was placed. Postoperative transthoracic echocardiography (TTE) revealed that pericardial blood volume was significantly improved with only a small amount left anteriorly. The pericardial thrombus was no longer evident. She remained in the hospital for two days after the procedure. The drainage catheter was removed on the second day when it drained less than 25 ccs of fluid over the day. She was subsequently discharged in a stable condition, and an outpatient follow-up was advised. |
R B, a 15-year-old girl, came to our unit for evaluating facial paralysis on the right side. As reported by her mother, the anamnesis was totally negative not describing any important diseases, and drugs assumption and family occurrence of this condition as well as previous episodes of facial paralysis were also excluded.\nThe patient reported that the symptoms appeared the day before the medical examination and consisted in acute pain on the right side of the face, difficulty in biting, dripping saliva from the mouth right corner, and impossibility to obtain a complete closure of the left eye. The clinical observation revealed a facial asymmetry with edema in the right jaw, difficulty of facial movements on the right side when smiling, and asymmetry of the eyes. Due to these aspects, the diagnosis was established as an idiopathic facial paralysis, and according to the House Brackmann scale, it was classified as IV degree (moderate severe dysfunction) ().\nIt was decided to treat the disease with an “self-administered PBM,” and the patient received the device as well as the instructions to correctly use it. The same day, the patient started the treatment applying the laser device (B-Cure Laser Pro, Good Energies Ltd., Haifa, Israel) on her own; this device is a class II laser according to ANSI classification, emitting in the near IR portion of the spectrum (808 nm) with a green LED aiming beam for indicating the irradiated zone of 4.5 cm2; its output power of 250 mW is emitted in micropulses with a frequency of 15 kHz (energy per minute of 14.4 J and fluence per minute of 3,2 J/cm2).\nTreatment was performed twice a day by cutaneous applications, each 15 minutes (total fluence: 48 J/cm2) by putting the device in contact with the skin of the right side in the area corresponding to the parotid gland, as reported in other different case reports []. |
A 53-year-old Caucasian woman was referred to our institution because of the suspicion of peritoneal carcinomatosis, raised by the findings of ascites at a transvaginal ultrasound performed as a yearly routine exam; a pre-surgical staging exam with computed tomography (CT) scan show thickening of the gastric walls, multiple omental nodules and ascites ().\nHer previous personal history was unremarkable and she denied any clinical symptom or cancer history.\nAt our hospital, she underwent an esophagogastroduodenoscopy and colonoscopy, with results negative for gastric/colon cancer.\nHer comprehensive metabolic profile revealed mild liver dysfunction with an alanine transaminase of 77 U/L and aspartate transaminase of 71 U/L. When tumour markers were assessed, CA125 demonstrated increased levels of 290 U/mL (normal values <35 U/mL), whereas carcinoembryonic antigen, CA 19.9 and other immunohistochemical markers were within the normal ranges. Serological assessment of HIV, hepatitis C virus and hepatitis B virus were negative.\nTen days later, the patient underwent an ultrasound-guided biopsy () with a diagnosis of suspicious carcinoma from the an unknown primary site.\nAfter 2 weeks, the patient received a CT scan of the thorax (to complete pre-operative staging), demonstrating a spontaneous (with no therapy) dimensional and numerical reduction of peritoneal lesions in the upper abdomen, partially included in the chest CT scan, as well as resolution of peri-hepatic and peri-splenic ascites ().\nSince there was no evidence of primary cancer at pre-operative examinations and the second CT scan revealed a partial resolution of peritoneal implants and ascites without therapy, the suspicion of an infectious disease was raised.\nThe pathological evaluation of the biopsies performed on the omentum and peritoneum revealed the presence of lymphoid aggregates with a central core of epithelioid cells with large eosinophilic cytoplasm, without atypia or mitosis () and with no immunohistochemical marker of oncologic malignancy. Due to the presence of necrotic nodules with histiocytes and giant cells, a Ziehl–Neelsen stain was performed to identify bacilli (), whose presence was then confirmed with molecular assays.\nBecause of the uncertain result of the biopsy and the conflicting results of the 2 CT scans, in order to rule out malignancy with certainty, the patient underwent a laparoscopic surgery in 2 weeks.\nDefinitive histological diagnosis excluded the presence of malignant cells and reported a necrotising inflammation caused by non-tuberculous mycobacteria. Hence, the patient was sent to a hospital with expertise in infectious diseases.\nA follow-up CT scan performed 1 year later confirmed a complete recovery of peritoneal findings. |
Due to a positive family history of esophageal cancer and episodes of heartburn, a 66-year-old male underwent an endoscopic gastric examination in 2013, where gastritis was suspected. Furthermore, a small mucous tongue was revealed at the level of the diaphragmatic opening suspicious for Barrett’s esophagus. The histological examinations revealed type C gastritis, so no Helicobacter pylori eradication treatment was necessary. Due to intermittent heartburn and regurgitation, treatment with a proton pump inhibitor (PPI) was implemented.\nOn a chest CT, which was conducted in November 2015 to investigate a chronic cough, a long segment widening of the esophagus was described as an incidental finding without evidence of tumor. On endoscopy, once again, there was no evidence of dysplasia. Following esophageal manometry, achalasia was suspected. With long-term treatment in the form of a PPI, the patient was free of complaints. Therefore, the patient had GERD-like symptoms (gastroesophageal reflux disease), and he also suffered from a hiatus herniation with moderate reflux disease, which was effectively better treated with PPI. These circumstances made it hard to establish the diagnosis. A follow-up examination with endoscopy was conducted in 2016. At this point, the patient did not complain of dysphagia and had hardly any other symptoms or findings.\nIn August 2018, a barium swallow examination was performed as a follow-up exam. This showed regular oral and pharyngeal phases of deglutition. The upper esophagus was not distended and exhibited a proper mucosal surface ()). A harmonic dorsal impression of the esophagus due to prominent Arteria lusoria was demonstrated at the level of the aortic arch. (). During deglutition, the retrocardiac esophagus was dilated by 4 cm. In addition, an air-fluid level, delayed peristalsis, and tertiary contractions could be seen (). Three centimeters above the diaphragm, a harmonic dorsal impression with ventral dislocation of the esophagus could be demonstrated. The length of compression of the esophagus measured 2.5 cm, with a prominent contrast filling defect (). Distally, the esophagus presented a regular diameter, and the cardia showed a regular opening up to 2 cm with a regular mucosal surface. There was normal mucous folding of the stomach and rapid gastric passage into the duodenum. There was no constant sphincter opening typically seen in patients with scleroderma and no signs of achalasia; the mucosal surface was normal with no signs of cancer.\nThe chest CT images from 2015 retrospectively revealed the cause of the esophageal impression: the esophagus was pinched between the left ventricle and aorta. The aorta crossed the spine right at that level, which caused compression of the esophagus ().\nFor further clarification, another esophagogastroduodenoscopy was conducted. This examination showed a normal z-line and no signs of reflux esophagitis (). Furthermore, a small axial hiatal hernia was detected. The dilation of the esophagus could be confirmed, matching the barium swallow. The distal esophagus demonstrated dorsal bulging with arterial pulsation without mucosal damage. The endoscope easily passed this aortic narrowing of the esophagus into the normal stomach ().\nDue to good esophageal patency, no further treatment was necessary. With regard to the positive family anamnesis of esophageal cancer, a follow-up of alternating barium swallow and esophagogastroduodenoscopy are recommended at intervals of one to two years. |
A healthy Caucasian 28-year old female was referred to our dental clinic with a continuous dull pain in the left mandibular region, which had started after initiation of root canal treatment on the first molar by her general dentist about two weeks before. Upon clinical examination, a localized inflammation overlying the buccal mucosa in furcal region of the tooth #36 became evident which was tender on palpation. The tooth had no tenderness on percussion and no mobility. Careful periodontal probing showed a pocket depth within the normal range (<3 mm). Radiographic examination revealed overextended endodontic access cavity preparation and extensive destruction of pulp-chamber floor ().\nIn the radiography a small piece of amalgam was evident in the apical extend of the perforation which is believed to have separated from the previous amalgam restoration of the tooth. The location of this small piece was an indicator of wide destruction of the interradicular bone during the preparation of the access cavity ().\nThe possible treatment options, including tooth extraction with/without replacement, tooth hemisection and non-surgical perforation repair with subsequent continuing of the root canal therapy (RCT) and coronal restoration were explained for the patient. The patient preferred the option of saving the tooth via a non-surgical endodontic treatment and furcal perforation repair with CEM cement. She also read and signed an informed consent.\nBefore the start of the treatment, mouth rinse with 0.2% chlorhexidine rinse (Behsa Co., Tehran, Iran) was carried out in order to control the oral microbial flora. After administering local anesthesia (2% lidocaine with 1:80000 adrenalin; Daroupakhsh, Tehran, Iran) and proper tooth isolation, the temporary restorative material was removed with a slight correction in the outlines of access cavity and then all canal orifices were located. The cavity and perforation site were copiously irrigated with full concentration of sodium hypochlorite and then normal saline. All canal orifices were preflared with #2 and 3 Gates-Glidden drills. Then the canals were blocked with appropriate-sized gutta-percha points to avoid obstruction with perforation repair material. CEM cement (BioniqueDent, Tehran, Iran) was prepared according to manufacturer's instruction and was placed into the cavity. In order to obtain a good marginal adaptation, the bulk of biomaterial was gently packed with a dry cotton pellet. After a few minutes required for initial setting of CEM, the gutta-percha points were removed and the biomaterial was covered with a moistened cotton pellet. After application of temporary restoration (), the patient was dismissed.\nThe second visit was a week later during which the patient was clinically evaluated. All the sign/symptoms had subsided and the tooth underwent conventional non-surgical root canal treatment. The working length was established and all four canals (naming distobuccal/lingual and mesiobuccal/lingual) were mechanically shaped using modified step-back technique and chemically cleaned by means of copious irrigation with 5.25% sodium hypochlorite and then normal saline. The root canals were filled with gutta-percha points (Ariadent, Tehran, Iran) and Roth root canal sealer (Roth's 801, Elite Grade; Roth Int., Chicago, IL, USA) using lateral condensation technique; the tooth was then restored with amalgam in the very same session. After taking the postoperative radiography (), the patient was put on a scheduled follow-up.\nDuring the one year follow-up, the tooth remained functional and asymptomatic. Clinical examination showed that the tooth had no tenderness to percussion/palpation and the probing depth remained within normal level. Radiographic examination demonstrated adequate filling and sealing of the perforation site with normal periodontal apparatus (). |
A 50-year-old man who visited the hospital every year after endoscopic submucosal dissection of early gastric cancer underwent a chest computed tomography (CT) for cancer screening. The patient was a 50-pack-year current smoker, but he did not have any respiratory or systemic symptoms. The chest CT scan showed small nodules on both sides of the lungs. There was a peripherally located small nodule with lobulation in the apicoposterior segment of the left upper lobe (LUL) and two tiny nodules in the posterobasal segment of the right lower lobe (RLL) (Figure ). Levels of tumor markers, such as carcinoembryonic antigen and cancer antigen 19-9, were within the normal range. The preoperative forced expiratory volume in 1 s was 3.02 L (80% of the predicted value) without an obstructive pattern. Given that the patient was a current smoker, was older than 50 years, and had a gastric cancer history, we decided to perform a surgical biopsy of the nodules.\nUnder general anesthesia, the surgeons performed a wedge resection by VATS. The LUL nodule was palpable to the finger and was 1 cm in size, hard, and capsulated in nature. There was no adhesion with the lung parenchyma or pleura. The frozen biopsy showed a hypocellular nodule in a fibrotic stroma. It was reported as a fibrotic nodule. The RLL nodules were also considered as benign because of similar features. A CT follow-up of these nodules was planned rather than surgical resection. Postoperatively, the patient did not show any surgical complications.\nThe pathological findings of the resected LUL nodule indicated a well-demarcated hypocellular hyalinized nodule. Generally, the neoplastic cells were loosely embedded in hyalinized fibrous stroma individually, although it was more dense cellularly at the periphery of the nodule. The neoplastic cells showed no nuclear atypia and contained variably prominent eosinophilic cytoplasm with or without cytoplasmic vacuoles which was reminiscent of endothelial differentiation. The tumor cells also showed immunoreactivity for CD31 (Figure ). These features are consistent with the histological characteristics of epithelioid hemangioendothelioma [,]. We decided to perform consecutive surgery of the two remnant tiny RLL nodules. Given that the preferred locations of epithelioid hemangioendothelioma are the liver and soft tissue, we performed an abdominopelvic CT before the second operation. There was no definite focal wall thickening and no enhanced masses were detected in the intraabdominal organs. The second operation was performed successfully using VATS; the pathological findings of the two tiny nodules were similar to those of the previous LUL nodule. After the second surgery, we performed follow-up chest CTs at 6-month intervals. There has been no evidence of recurrence or other metachronous nodules for 18 months. |
Our patient is a 63-year-old male who underwent heart transplantation for stage D heart failure with reduced ejection fraction (HFrEF) due to ischemic cardiomyopathy. The patient's postoperative course was unremarkable, and there was no evidence of allograft rejection on the 12 biopsies obtained within first six months posttransplantation. He presented for an outpatient right heart catheterization (RHC), coronary angiography, and EMB for his routine one year posttransplantation evaluation. Coronary angiography revealed a very large RCA measuring 5 mm with a fistulous connection from posterolateral branch to possibly the right atrium (Supplemental ). There was no “step-up” in venous saturations suggestive of an intracardiac shunt. Hemodynamics were unremarkable with a pulmonary artery pressure of 29/18, a pulmonary capillary wedge pressure of 12 mmHg, and a cardiac output of 5.6 L/min. The patient's transthoracic echocardiogram (TTE), two months posttransplant, was suggestive of a very subtle right coronary artery to right atrial shunt which was not noted on the initial read (Supplemental ). This anomaly was not present on the donor heart TTE immediately pretransplant. A retrospective systematic review of all EMB slides revealed a blood vessel sample present on one of the prior biopsies (). This particular biopsy was obtained via a transjugular venous access and under fluoroscopy guidance. We also pursued a coronary CTA that revealed an ectatic RCA measuring approximately 6 mm in diameter and an ectatic posterolateral branch measuring 4.5 × 5.5 mm with a fistulous communication to a dilated coronary sinus (Supplemental Videos ). The fistula was not hemodynamically significant as the patient had continued to remain asymptomatic, with normal right atrial and right ventricular dimensions and normal filling pressures on RHC. Hence, the patient did not undergo any interventions for this fistula. Despite this anomaly, the patient remained asymptomatic with improving exercise capacity on a 6-month follow-up from his heart angiogram. He was managed conservatively with good follow-up. |
A 21-year-old female patient came to our service with a complaint of unilateral right-onset headache associated with diplopia initiated 6 months earlier. She had no personal or family remarkable antecedents. She never smoked. Six months earlier, the patient started to experience one-sided right throbbing headache. She denied nausea, vomiting, or photo- or phonophobia. Fifteen days after the pain onset, she noticed double vision and medial deviation of the right eye, which forced her to wear an eyepiece to perform her activities and drive. She went to several centers and used various medications such as paracetamol, nonsteroidal anti-inflammatory drugs (NSAIDs), opioids, and triptans without improvement. Three months earlier, she started using dexamethasone 4 mg daily with partial pain control but maintenance of diplopia.\nAt the examination, the patient had cushingoid face, violaceous striae, and right VI cranial nerve palsy with no other neurological changes. Blood tests were normal (). A contrast-enhanced MRI scan of the brain did not show any remarkable features (). A spinal tap released crystalline cerebrospinal fluid (CSF) with an opening pressure of 14 cm of water. Biochemical, microbiological, and cytological analyses of the CSF were normal (). CT scan of thorax did not show any evidence of lymphoma or sarcoidosis.\nPrednisone 1mg / kg was then started. With one week of treatment, complete reversal of ocular paralysis and remission of pain were observed. However, when the corticoid was gradually withdrawn, the patient returned to pain and returned to paralysis of the VI right pair. The prednisone was increased again to 1 mg / kg this time with reversion of ocular paralysis but without pain control. Several prophylaxis attempts were made with beta-blockers, calcium channel blockers, topiramate, and tricyclics without any symptomatic control that would allow corticosteroid withdrawal.\nThe pregabalin 150mg daily was then introduced. With 7 days of medication onset there was already an important remission of pain. With 15 days of pregabalin initiation, the retitration of prednisone was started without any intercurrence and the patient reversed the exogenous Cushing syndrome. Pregabalin was maintained for one year and retracted. Currently, the patient has been free of pain for 2 years. |
A 44-year-old man had been treated with peritoneal dialysis for chronic kidney disease stage-V (CKD-V) for more than 6 years. The ESRD was caused by chronic nephritis. He has a history of hypertension for 6 years without other particular disease. He stated had edema of eyelid and low limbs intermittently and weakness of whole body. A peritoneal dialysis catheter (a right side straight two cuffed Tenckhoff catheter) was placed in August 2012, and the patient received peritoneal dialysis regularly since that time. Several episodes of peritonitis caused by peritoneal dialysis lasted more than 4 weeks only with symptomatic and empirical treatment. In May 2019, Leakage of peritoneal dialysis fluid was noted at the exit-site of the PD catheter. Three days later, the patient developed chills and fever and diagnosed with peritonitis (Supplementary Table ). After a week of antibiotic therapy, (Imipenem 0.5 g Intraperitoneal for 4 h once, Meropenem 0.5 g Intravenous infusion Q12h), his body temperature returned to normal; however, the peritoneal dialysis effluent became turbid, and passage of watery stool occurred immediately after each infusion of peritoneal dialysate into the abdominal cavity. The patient had no abdominal pain or distention.\nAbdominal computed tomography (CT) revealed inflammation in the abdominal cavity, extensive peritoneal calcification, and appropriate positioning of the peritoneal catheter, but intestinal perforation was not evident (Fig. ). Peritoneal dialysate containing methylene blue reagent was injected into the abdominal cavity. After 2 hours, anal drainage was light blue (Fig. ). Thus, an intestinal fistula was suspected. Colonoscopy revealed methylene blue at the area of the appendiceal orifice (Fig. ), which confirmed the presence of a communication between the abdominal cavity and the bowel lumen. Peritoneal dialysis was discontinued and hemodialysis was initiated. The peritoneal catheter was removed by open surgery. No abdominal pain, abdominal distension, or other symptoms occurred during the follow-up period, which lasted a minimum of 3 months. |
A 61-year-old woman presented with two supernumerary nipples located along the milk line on each side of the upper abdomen. During a few months before referral, the patient had noticed a firm palpable mass in close relations to the supernumerary nipple on the right side (). She had no other symptoms. Bilateral mammogram and ultrasound revealed normal breast parenchyma. Ultrasound of the supernumerary nipple on the right side confirmed a small mass in relation to this nipple, presenting as a hypoechoic, well-defined area, measuring approximately 10 mm in diameter ().\nUpon clinical examination, the supernumerary nipple on the right side presented with inversion and a palpable firm mass underneath this nipple. Ultrasound-guided needle core biopsy (16G) was performed (). The pathology report described elongated epithelial islands composed of cubic cells, with a small centrally located lumen with focally identifiable cuticles. The epithelial structures were surrounded by a dense fibrous stroma. The microscopic analysis indicated the possibility of SyT and the lesion was categorized as a B3-lesion (a lesion with uncertain malignant potential) ().\nFollowing a Multidisciplinary Team Conference, it was decided to recommend resection with a 5 mm rim of normal tissue, which was subsequently performed ().\nMacroscopically the tumour measured 7 x 5 x 5 mm and was described as a firm and grey-white tumour, in close relations to the supernumerary nipple. Microscopically the tumour measured 13 mm in diameter and was localized in the deep part of dermis and underlying subcutaneous tissue with no relation to the epidermis. The tumour was composed of solid trabecular and glandular imitating formations with focal cysts. The epithelium consisted of cells with slightly irregular nuclei and indistinct nucleoli, surrounded by an eosinophilic cytoplasm. The glandular structures were lined by cuboidal cells. The tumour was in close association with the lactiferous ducts and smooth muscle of the nipple (). Foreign body giant cell reactions, due to ruptured cysts, were identified.\nImmunohistochemical analysis showed positive reaction for CK5, CK14, and P63 () in association with the epithelium presenting as solid cords, whereas the glandular luminal cells showed positive reaction for CK7 (). Thus, the immunohistochemical analysis demonstrated the complex nature of this lesion. The diagnosis was SyT based on the pathology report.\nA re-excision was performed due to insufficient rim of normal tissue in the cranial direction in order to minimize the risk of recurrence. |
An 80-year-old Caucasian female with history of hypertension and chronic back pain presented for emergent repair of a 7.2 cm aneurysm of the ascending aorta with Stanford classification type A dissection. Because the ascending aorta was unsuitable for arterial cannulation, the surgeon elected to perform axillary cannulation via the right subclavian artery with side graft anastomosis. The patient arrived to the operating room (OR) with nicardipine and esmolol infusions running through an 18-gauge peripheral intravenous (IV) line in the right antecubital (AC) fossa. Prior to induction of anesthesia, we disconnected the infusions from the right AC and administered medications though an 18-gauge IV in the left forearm. The patient also had a left radial arterial line (AL), and we placed the pulse oximeter and noninvasive blood pressure (NIBP) cuff on the RUE. After intubation, we placed a right radial AL, and the surgeons placed a left femoral AL. All arterial pressures correlated closely. Additionally, a 9-French central line was placed in the right internal jugular vein. The patient was cleansed and draped for surgery with her arms tucked to her sides.\nShortly after the procedure began, the right radial AL tracing went flat, and the pulse oximeter waveform was lost. We attributed this to the surgeon partially clamping the right subclavian artery in preparation for arterial cannulation. We switched the pulse oximeter to the left hand and relied on the left radial and femoral AL for pressure readings. Just prior to arterial cannulation, we noted that the right radial pressure returned, although about 20 points lower than the left radial/femoral. Immediately after initiating CPB, the right radial mean arterial pressure (MAP) increased to 200 mm Hg, and left radial/femoral MAP decreased from 60 to 30 mmHg. The perfusionist alerted the surgeon about the high line pressures and decreased CPB flows. After a brief attempt to troubleshoot and adjust the cannula with little improvement in pressure or flow, the surgeon proceeded with the operation. Over the next few minutes, the left radial/femoral MAP increased to 60 mmHg.\nAs deep hypothermic circulatory arrest (DHCA) was initiated, the right radial MAP decreased to 30 mmHg and left radial/femoral MAP decreased to 10. When CPB was reinstated, right radial MAP again increased to 200 mmHg. After 29 minutes of DHCA and 265 minutes of CPB, the patient was successfully weaned from CPB, and right radial MAP decreased to about 10 points lower than left radial MAP.\nDespite these issues, the surgery was otherwise uneventful. However, upon the surgical drapes being taken down, we noticed that the patient's RUE was swollen with blisters and bullae from the shoulder to the hand; yet, the skin of the upper arm where the NIBP cuff had been placed was normal as seen in figures. The IV in the right AC appeared to be infiltrated and weeping fluid even though we had not used it during the case and did not have any IV fluids attached to it (Figures and ). The IV and right radial AL were removed in the OR, and a Xeroform gauze dressing was applied to the RUE with the surgeons present. The intensive care unit nurses were instructed to elevate the arm and perform hourly neurovascular checks.\nOn postoperative day 1, the patient complained of tenderness and burning in the RUE, but she maintained adequate capillary refill, motor function, and sensation. Plastic Surgery was consulted to rule out compartment syndrome. They were unsure of the diagnosis but recommended nonoperative management and continued neurovascular checks. Eventually Dermatology was also involved, and they performed a punch biopsy of the patient's right dorsal hand. Their initial diagnosis was allergic contact dermatitis (ACD) due to the fact that the area of skin covered by the NIBP cuff was spared. However, the biopsy showed pauci-inflammatory dermal-epidermal blistering, which did not favor ACD. Direct immunofluorescence was also negative, ruling out localized pemphigus. Given the histologic findings, the final diagnosis was hydrostatic edema/bullae correlating with rapid edema during surgery. The patient continued to be managed nonoperatively with 1% triamcinolone ointment and gauze dressings, and within one month the blisters had completely resolved. |
The patient was a 21-year-old woman, with an unremarkable family medical history. She had first undergone treatment for GISTs associated with an incomplete-type Carney’s triad 11 years prior, at the age of 10 years.\nAt the time of her first admission and treatment, the patient had presented with black stools as her chief complaint. Endoscopic examination revealed the presence of two submucosal tumors in the pyloric antrum (Fig. ) []. Magnetic resonance imaging showed multiple nodular tumors presenting as low signals on the T1-weighted image and high signals on T2-weighted image of the gastric body to antrum. In addition, computed tomography showed pulmonary nodular shadows in the S1 region in the right lung and lingular region in the left lung (Fig. ) []. Based on these findings, the patient underwent open partial gastrectomy with Billroth-I reconstruction. Histopathological examination showed a positive result for c-kit and negative for S-100 protein; thus, we provided a diagnosis of wild-type GISTs. There was no vessel or lymphatic invasion, and MIB-1 labeling index was below 10%. The GISTs were diagnosed to be of high risk because the pulmonary tumors were considered to be metastatic. Two months after this gastric surgery, imatinib was initiated for the treatment of the pulmonary tumors. As the pulmonary tumors grew slowly, the type of TKI was changed from imatinib to a combination of sunitinib and regorafenib. This change did not lead to a positive tumor response. As the pulmonary tumors in the right upper lobe increased in size, a right upper lobectomy was performed, 9 years after the index gastric surgery. The tumor was uncoated and displaced the surrounding lung tissue (Fig. ) []. Histopathological examination of the resected tumors confirmed the diagnosis of pulmonary chondroma. There was no vessel or lymphatic invasion. Based on the pathological result of pulmonary chondroma, a further histopathological re-evaluation of the resected gastric specimen was conducted that revealed a succinate dehydrogenase (SDH) deficiency of the tumors. Based on these findings, a diagnosis of an incomplete-type Carney’s triad was provided. The patient was followed up with yearly ultrasound examinations, with no indication of recurrent tumors in her stomach over the short-term follow-up.\nHowever, 2 years after her right upper lobectomy, at the age of 21 years, new gastric tumors were observed with ultrasound examination. Endoscopic examination of the upper gastrointestinal tract revealed the presence of multiple submucosal tumors on the residual side of the lesser curvature (Fig. a, b). Biopsy confirmed a pathological diagnosis of GISTs, with SDH deficiency. On computed tomography imaging, multiple masses were observed in the whole stomach, growing into the inner cavity. There was no obvious disseminated involvement nor metastatic lesions (Fig. ). The patient was referred to our center for further assessment and treatment.\nThere was no evidence of extra-adrenal paraganglioma. A clinical diagnosis of recurrent GISTs, as a component of an incomplete-type Carney’s triad, was made. We decided to proceed with laparoscopic examination and treatment, as appropriate. On laparoscopic examination, further multiple nodules were observed on the serous surface of her stomach, with mild adhesion around the gastroduodenal anastomosis that could be easily divided (Fig. a). Based on these findings, we proceeded with completion gastrectomy, with D1 plus lymph node dissection and Roux-Y reconstruction, performed laparoscopically (Fig. b). Macroscopic examination revealed that the sporadic gastric stromal tumorlets were multifocal subserosal polypoid nodules (n = 8), with the largest being 52 × 30 × 25 mm in size (Fig. ). The tumor cells showed an epithelioid pattern, and all eight tumors were diffusely immunoreactive on CD117 and CD34 antibody assay (Fig. a–c). Vessel or lymphatic invasion was not observed. The Ki-67 index was 10%. Tumors were SDH deficient, confirming the diagnosis of GISTs as a component of an incomplete-type Carney’s triad. We did not observe any benefit of chemotherapy on the harvested tumor specimen.\nThere was no complication after surgery, and the patient’s postoperative course was uneventful. She was discharged from the hospital in good condition and has been monitored carefully in follow-up, with no adjuvant chemotherapy treatment. Over the 7 months since the gastrectomy procedure, she has had no difficulty with oral intake, and there is no evidence of tumor recurrence. |
A 63-year-old Caucasian male presented with a 3-year history of ulcerating lesion involving the left cheek and parotid gland. Three years ago, he started to have slow-growing lesion arising from a nonhealing wound that occurred after a left cheek traumatic injury by a tree branch. The lesion was neglected until it started to increase rapidly in size over the last 5 months when he sought treatment. He reported associated left-sided hearing loss, weakness in eye closing, and mandibular weakness, all on the left side. He denied weight loss, trismus, or any lymphadenopathy.\nThe patient lived in California and had excessive sunlight exposure before he moved to Arkansas, where he received treatment. He has a past medical history of asthma, and cSCC of the right cheek required Mohs surgery. He reported no medications use or allergies. He denied tobacco, alcohol, or recreational drug use or any occupational chemical exposure. No history of immunocompromised status or immunosuppressive therapy reported. The patient’s mother died from colon cancer at age 46, and his brother had a history of melanoma.\nOn physical examination, there was an extensive bleeding fungating growth overlying the left cheek, extended to the left helix root and tragus [Figure –]. The lesion hung over the external auditory meatus, which was intact. The tympanic membrane was intact. There was no significant submandibular or cervical lymphadenopathy. He had pale conjunctivae. The remainder of his physical examination was unremarkable.\nThe patient was evaluated initially by a dermatologist, and his lesion biopsy showed poorly differentiated SCC. Given his extensive and high-risk SCC, he was referred to a head and neck surgeon. The computed tomography (CT) scan of the head and neck showed an extracapsular invasion in the soft tissues of the muscles of mastication and the zygomatic arch []. For the staging workup, the patient underwent a positron emission tomography/computed tomography (PET/CT) scan, which showed the giant invasive facial mass [] and interestingly showed an incidental finding of a 2.1 cm left cortical renal mass and bladder thickening [Figure and ]. The patient reported no history of gross hematuria or flank pain. On the basis of his staging workup, he was considered to have a T3, N0, and MX poorly differentiated SCC.\nThe patient was scheduled for an elective surgical resection and a reconstruction flap. His preoperative evaluation revealed increased fatigue and dyspnea with exertion, palpitations, dizziness, and lightheadedness without syncope. His preoperative laboratory data showed hemoglobin of 5.3 g/dL, mean corpuscular volume (MCV) 97.1, with normal B12, folic acid, and iron studies. He reported a long-term intermittent low-volume oozing from the facial lesion, which increased to an intermittent frank bleeding over the last 3 months. He denied hematochezia, hematemesis, hematuria, or coffee-ground emesis. Despite blood transfusion of two packed red blood cells (RBCs) units in the outpatient settings, his hemoglobin improved only to 6.5g/dL. Due to his blood loss anemia, he was hospitalized preoperatively and received another unit of packed RBCs for medical optimization before his planned surgery.\nHe underwent an excision of the left zygomatic bone, left total parotidectomy with facial nerve sacrifice, and selective left neck dissection levels of I–IV []. The reconstruction of the left facial defect was performed with an anterolateral thigh free flap with microvascular anastomosis [Figure and ]. Pathology studies showed deeply invasive squamous cell carcinoma invading the parotid gland. All the margins and excised lymph nodes were negative for cancer.\nUpon his postoperative follow-up, he was, expectedly, noted to have iatrogenic Bell’s palsy. His ophthalmology examination revealed no corneal epithelial defect and he was started on high-viscosity artificial tears. The radiation oncology team recommended adjuvant radiation therapy to the deeper structures of the operative site and he is scheduled to undergo an intensity-modulated radiation therapy (IMRT) to preserve salivary function and minimize the risk of flap failure. For his renal mass, the patient was evaluated by urology and based on the peripheral location, renal cell carcinoma was suspected. He is scheduled for percutaneous CT-guided cryoablation after recovery from the facial reconstructive surgery. |
A 72-year-old male, 70 kg and 167 cm height, who underwent L2–L5 decompression and transforaminal interbody fusion presented with chief complaints of pain in the lower back and bilateral lower limbs for the past 1 month. During preoperative anesthesia workup, it was found that patient was on irregular medications for hypertension. The preoperative blood investigations were within normal limits, and electrocardiography showed normal sinus rhythm. As a part of the protocol in our hospital, echocardiography was done and there was no regional wall motion abnormality seen and ejection fraction was 55%. Premedication was given to the patient, and antihypertensives were continued as per schedule. Standard general anesthesia was given. Routine monitors (electrocardiogram, noninvasive blood pressure, pulse oximetry, and capnography) were attached, and intravenous glycopyrrolate 0.2 mg was given. Induction of anesthesia was done using titrated doses of propofol, and muscle relaxation achieved using rocuronium 50 mg. Intravenous morphine 7 mg was used to provide analgesia. Intubation response was prevented using fentanyl 100 mcg. The maintenance of anesthesia was carried out using oxygen/air mixture and sevoflurane along with intermittent boluses of atracurium. Volume control mode of ventilation was used intraoperatively, and a target end-tidal carbon dioxide of 32–35 mmHg was kept. Vascular cannulation in the left radial artery was performed for continuous blood pressure monitoring. In addition to the routine monitors, temperature and urine output were monitored. In order to facilitate surgery, after induction of anesthesia patient was made to lie in the prone position. Postprone positioning, all pressure points including eye pressure were checked, the neck was kept in the neutral position, vitals were stable, and surgery was started. During the middle of surgery when transforaminal dilatation near the lumbar vertebra (L3) was being performed, there was a sudden onset of bradycardia (heart rate (HR) dropped to 38 beats per min) with a simultaneous fall in arterial blood pressure (72/34 mmHg), with no change in end-tidal carbon dioxide levels and oxygen saturation []. The surgeon was informed about the event, and the surgical stimulus was released immediately. Within few minutes following the release of surgical stimulus, HR and arterial blood pressure came to baseline. There was a recurrence of similar episodes twice during the surgery which again responded to the removal of surgical stimulus. Towards the end of surgery, the patient's trachea was extubated in the supine position after reversal of neuromuscular blockade once the patient became fully awake, conscious, and was following commands. Furthermore, the patient's stay in the postoperative anesthesia care unit was uneventful and there was no neurological deficit. Consequently, the patient was shifted to the ward for further management. |
A healthy looking, 50-year-old Nepali man came to our clinic with a complaint of multiple growths on his scrotum for 15 years. The growths started as a single lesion on the right side of his scrotum, with the gradual appearance of similar lesions on other parts. Several of these lesions coalesced at various places to form large-sized nodules. The condition was mostly asymptomatic with an occasional complaint of itching. There was no history of pain, burning sensation, trauma, ulceration, or discharge. The lesions did not interfere with urination or sexual activities. He was worried because of the increasing size of the growth and hence came to us for advice. He did not give a history of any systemic illness including metabolic, autoimmune, or malignant disorders. There was also no history of a similar complaint in his family.\nOn physical examination, multiple pink to brown nodules ranging in size from 0.5 × 0.5 × 0.5 cm to 3 × 3 × 1 cm involving almost half of his scrotum were noticed (Fig. ). The skin over the nodules was shiny with several yellowish points indicative of underlying calcium deposition. The skin surrounding the nodules, testis, and penis was normal on palpation. The nodules were painless and firm in consistency. On laboratory examinations the following were found to be within normal limits: serum calcium, phosphorus, parathyroid hormone, and vitamin D hormone levels; uric acid; alkaline phosphatase; and lipid profile. Based on clinical features and laboratory reports, a diagnosis of ICCS was made.\nHe was advised a scrotectomy under spinal anesthesia, which he refused. The nodules were excised under local anesthesia in several sittings. The skin was sutured using chromic catgut (4–0). His postoperative period was unremarkable with good cosmetic result and no evidence of recurrence in a 1-year follow-up period. The cut section of nodules showed solid white to yellow homogenous areas. Histopathological examination revealed skin tissue lined by keratinized stratified squamous epithelium. The underlying dermis had areas of fibrosis and calcification (Fig. ). Numerous multinucleated giant cells were also seen (Fig. a and b). An obvious cystic structure was absent. |
A 34-year-old male patient with no prior medical history presented at the Emergency Department reporting ingestion of several metallic objects one hour before. The clinical examination was unremarkable, and the patient was in overall excellent condition. X-ray studies revealed at least 4 metallic objects in various parts of the GI tract ().\nConsidering the timing of ingestion and the type of the ingested materials, endoscopic extraction was decided. Esophagogastroduodenoscopy (EGD) was performed approximately 2 hours after the ingestion. Four objects were visualized at the time of endoscopy (one screw in the first part of the duodenum, one elongated pin impacted in the antrum wall, one coin, and one small metal scrap in the stomach). The coin was easily removed from the stomach using a Roth Net. An overtube was then introduced in order to safely retrieve the remaining 2 larger sharp objects. None of the remaining objects could fit in the overtube, and the patient's tolerance to the procedure was poor despite maximal sedation. No other protective devices such as rubber hoods were available at the time of endoscopy. Therefore, any further efforts to extract the objects were abandoned, and the patient was returned to the surgical ward for monitoring. Recognizing that complication rates could be high in this patient and that referral to another better equipped facility would require time, such that it would expose the patient to the risks of delayed intervention, we improvised a handmade rubber hood, constructed from a plastic transfusion pressure infusor (). This material was chosen because it was flexible enough to invert back on itself in the cardia upon withdrawal of the scope, yet thick enough to withstand puncturing. The hood was fastened with silk tape to the distal end of a 9.8 mm gastroscope (GIF-Q165 Olympus Corporation, Tokyo, Japan), in a coned fashion (to facilitate the entrance of the foreign body into the bell of the hood), and then inverted (Figures and ). A test was carried out before scope insertion to make sure that the foreign body fits in the hood ().\nThe patient was called for the second EGD three hours after the first one. The two larger foreign bodies were still in the stomach, whilst the small metal scrap had migrated distally. With the help of mild sedation (3 mg of midazolam), the pin and the screw were captured using rat-tooth forceps, pulled into the hood, and extracted (). The hood inverted back on itself as expected, with no tissue injury upon second endoscopic look. The procedure lasted only 15 minutes with excellent tolerance from the patient.\nThe patient was returned to the ward for monitoring because two sharp objects were still in his small bowel. Repeat X-ray studies the following days confirmed successful discharge of the foreign bodies per rectum without any complications. |
A 56-year-old man who had diabetes mellitus and a recent diagnosis of prostate cancer presented with acute abdomen to the emergency room about two hours after having received endorectal prostate MRI examination. Prior to the event, he had been initially referred to our hospital because of one-month history of gross hematuria. Serum prostate-specific antigen (PSA) was 3.99 ng per milliliter. Examination of transrectal ultrasound-guided (TRUS) biopsy specimen of the prostate revealed prostate adenocarcinoma in the right anterior and posterior aspect with Gleason score of 9 (4 + 5). A whole-body bone scan reported bone metastasis to the thoracic spine (T11). Therefore, endorectal prostate MRI was arranged to evaluate the extent of cancer for the best treatment path. Patient had a light diet 1 day before and undergone cleansing enema early morning at the hospital prior to the procedure, while metal and electronic objects were removed. A 1.5 T endorectal MRI/MRSI was used (Fig. ). The endorectal coil was covered with latex condom to prevent contamination. During endorectal prostate MRI examination, patient maintained a left lateral decubitus position. The probe was lubricated with xylocaine jelly adequately and inserted through the anus to the rectum with mild force in cephalad fashion. Slight painful sensation of the lower abdomen was experienced momentarily but soon dissipated. The intrarectal balloon was inflated with 70 to 80 ml of carbon dioxide (Fig. ) to reach maximal image resolution. Patient tolerated the procedure well without immediate complications and was sent home afterwards. The MRI result revealed advanced prostate cancer with transcapsular invasion to periprostatic tissue, right seminal vesicle and urinary bladder, metastatic regional lymphadenopathies and multiple bony metastases.\nApproximately two hours following the examination, patient was seen in the emergency room because of a sudden onset of severe lower abdominal pain. A supine abdominal computed tomography (CT) showed diffuse pneumoperitoneum and perirectal extraluminal air with fat stranding (Fig. ), prompting an emergency exploratory laparotomy. Intra-operatively, a rectal perforation about 2 cm in diameter at the anterior wall of the upper rectum was identified, suggesting an iatrogenic relation during endorectal prostate MRI examination (Fig. ). A primary closure of the rectal perforation with sigmoid loop colostomy was performed. Nasogastric decompression was applied until the third day post-operatively. The intraoperative ascites culture result revealed ESBL-producing Escherichia coli infection. A concomitant urinary tract infection with Pseudomonas aeruginosa was reported. A colonoscopy was performed to evaluate the perforation site 10 days later. An approximately 8-mm residual perforation still presented in the anterior wall of the rectum, which was repaired by intraluminal endoclipping for perforation closure. The follow-up computed tomoraphy scan 20 days later showed absence of intra-abdominal free air. A good healing process of the perforation was seen during the colonoscopic examination. |
Case 1: A 21-year-old Japanese female. She was struck with lower-body paralysis of unknown origin at the age of 10, and has lived her daily life in a wheelchair ever since. In September 2012, she wore a new pair of leather shoes when going to work, thus resulting in pressure ulcers suddenly appearing the next day on the heel and first toe of her left lower extremity. Subsequently, the patient visited another hospital and underwent treatment with various topical ointments including dimethyl isopropylazulene and 0.9% iodine-containing ointment; however, the course showed no improvement for approximately 6 months, so she visited our hospital in April 2013. There were pressure ulcers measuring some 5 cm in diameter with an abnormal odor in the heel of the left lower extremity. With insufficient granulation, the ulcers had a defect extending to the subcutaneous tissue. Although the subcutaneous tissue was visible, the bones, tendons, and muscles were not exposed. A brownish-red eschar and a yellowish slough were attached to part of the wound floor, and the depth of the ulcers was partially indeterminable. Full thickness tissue loss had occurred. Subcutaneous fat may be visible but bone, tendon or muscles are not exposed. The case corresponds to National Pressure Ulcer Advisory Panel (NPUAP) Category stage III (). With the consent of the patient, oral administration of omega-3-acid ethyl esters (2 g once a day) was initiated while applying 0.9% iodine-containing ointment for the purpose of protecting the ulcer surface and the ulcers, which had not changed for 6 months, turned from a brownish-red color to a yellowish color at 2 weeks (, ), and thereafter entered the yellow phase at 4 weeks, while showing a contractive tendency (). At 8 weeks, the ulcer surface on the heel was further reduced, with progressed granulation in addition to a tendency for improvement. Moreover, in the ulcer on the first toe, epithelialization occurred around the wound, and the ulcer was reduced and improved entering the white phase (). At 10 weeks, the ulcer on the first toe healed completely (). However, diarrhea was caused as a side effect of omega-3-acid ethyl esters, and although continued oral administration was instructed with the aim of complete healing of the ulcer on the heel, it was discontinued in the end of September. |
A 62-year-old male with a past medical history of human immunodeficiency virus, who was receiving therapy, had a CD4 count of 641 and had been diagnosed with CMML 2 years ago presented with fatigue and dyspnea on exertion for 1 week. The patient had received different treatment regimens for CMML, including decitabine (5 cycles), ruxolitinib (8-month therapy) and azacitidine (2 cycles). He was recently started on hydroxyurea because of his disease progression and was sent for allogeneic hematopoietic stem cell transplant; however, he was deemed to not be a candidate because of renal dysfunction. The patient was admitted to the hospital for additional work up. He was found to have worsening anemia and thrombocytopenia as well as worsening leukocytosis (62 from 40 K 3 weeks prior to admission). He was diagnosed with autoimmune hemolytic anemia on this admission (positive Coombs test) and was started on prednisone. He received blood products, including several units of packed red blood cell and platelets, to maintain adequate hemoglobin levels and platelet counts. His bone marrow biopsy on admission showed dysmegakaryopoiesis and monoblasts accounting for 14% of the cellularity, which was consistent with CMML.\nThe patient was started on trimethoprim–sulfamethoxazole for Pneumocystis jiroveci pneumonia prophylaxis, hydroxyurea, IV fluids and allopurinol for tumor lysis syndrome prophylaxis. On Day 5 of admission, radiation oncology was consulted for palliative radiation for refractory and symptomatic splenomegaly because all medical therapy had failed. However, during the course of his admission, his respiratory status deteriorated slowly, requiring nasal cannula oxygen, and his X-ray showed evidence of bilateral infiltrate suggestive of pulmonary edema (Day 9 of admission). The patient’s echocardiogram was normal 2 months prior to his admission, and his current brain natriuretic peptide level was only 71 pg/ml; however, given his clinical and radiological status, he was started on IV Lasix. He continued to deteriorate despite diuresis and required noninvasive positive pressure ventilation. After pan cultures, he was placed on broad spectrum IV antibiotics, specifically vancomycin, piperacillin and tazobactam. He was intubated on Day 10 of admission. His X-ray and chest computed tomography 1 month prior to admission showed no evidence of opacity or acute infiltrate, and the abdominal section showed hepatosplenomegaly (, and ). A chest X-ray on admission () is shown. The chest X-ray () and chest computed tomography () on Day 8 show significant interstitial infiltrate. He continued to deteriorate despite full respiratory support and broad-spectrum antibiotics. His white blood cell (WBC) at the time of admission to the intensive care unit (ICU) was 124 000, and he expired after 6 h of mechanical ventilation. The patient experienced refractory multi-organ failure, and the ICU team was unable to perform bronchoscopy. Limited lung autopsy was requested, and the slides are shown ( and ). The final diagnosis made at autopsy was pulmonary leukemic infiltration associated with marked pulmonary congestion and hemorrhage. No evidence of infection was found in these cultures or on autopsy. |
Following a simple fall at home, an 85-year-old woman presented with back pain as a chief complaint and was referred to a hospital. Magnetic resonance imaging (MRI) taken two weeks after the initial onset showed a fresh L3 vertebral fracture although plain radiograms had failed to detect the fracture (). She initially had back pain without lower extremity symptoms and was treated conservatively. One month after the initial onset, bilateral leg pain appeared and gradually worsened. At four months after the initial onset she was referred to our hospital because the pain in her back and bilateral leg pain worsened in a standing or sitting position, decreasing during bed rest. The radicular leg pain was so severe that it was impossible for her to sit in a wheelchair. Physical examination revealed that bilateral radicular pain and numbness affected the bilateral anterior thigh, with MRC grade 3-4/5 weakness of the iliopsoas, and quadriceps femoris in both legs. Plain radiographs showed the L3 vertebral body more collapsed compared with the initial radiographs, and computed tomography (CT) scans showed a bony defect inside the fractured vertebral body (). MRI showed fluid collection in the fractured L3 vertebral body, suggestive of pseudoarthrosis of the L3 vertebral body. There was no retropulsion of bony fragments into the spinal canal. Spinal canal stenosis due to L4 spondylolisthesis was seen at the L4-L5 level (). However, these findings did not fully explain the patient's neurological status, which seemed to be caused by L3 (or L4) nerve root involvement. Importantly, bilateral foraminal stenosis at L3-L4 was present on MRI (), which may produce L3 nerve root compression.\nUpon the reexamination of the plain radiographs in standing and supine positions, we saw that the caudal part of the L3 vertebral body collapsed in the standing position, resulting in the exacerbation of the L3-L4 foraminal stenosis (). This led us to conclude that the patient's neurologic deficit was due to L3-L4 foraminal stenosis although a more detailed examination, such as electromyography, was not performed. Posterior decompression and fusion surgery decompressed the L3-L4 foramen and fused the unstable segment. The cranial half of the L3 lamina was removed and served as a local bone graft. The L3 pedicle was subtracted and the cranial part of the L3 vertebral body and L2-L3 disc were resected. Two intervertebral cages (PEEK OIC Cages: Stryker, Allendale, NJ, USA) with local and iliac bone grafts were placed between the L2 endplate and the remaining L3 vertebral body ().\nA pedicle screw system (Legacy: Medtronic Sofamor Danek, Memphis, TN, USA) was utilized, placing two screws above and two screws below the site, and posterolateral fusion was performed. A dramatic neurological improvement occurred following the surgery. By the 7th postoperative day, the patient could walk using parallel bars with a thoracolumbosacral orthosis for external support. No postoperative complications developed. Two months after surgery, the leg pain was completely gone, the muscle weakness fully recovered, and the patient could walk without support. At the final followup 18 months postoperatively she had no pathological symptoms related to spinal fracture. |
A 21-year-old male patient was referred to the department of conservative and endodontics after sustaining a complicated crown fracture on his permanent maxillary left central incisor in a road traffic accident, while riding a bike.\nClinical examination revealed complicated Ellis class III fracture of maxillary left central incisor with retained fractured segment and Ellis class II of maxillary right central incisor and lateral incisor []. Maxillary right central and lateral incisor showed no tenderness on percussion and no mobility. Intraoral periapical radiograph (IOPAR) was advised with respect to maxillary incisors. IOPA revealed an oblique crown fracture in tooth 21 [].\nProposed treatment to restore maxillary left central incisor included root canal treatment followed by flap refection and placement of biological post, made from the root cutting of extracted canine as well as the subsequent adaptation of patient's own crown fragment and direct composite restoration for maxillary right central incisor and lateral incisor. After agreeing upon the proposed treatment, a consent form was duly signed.\nFractured segment was removed from maxillary left central incisor [] and stored in artificial saliva until cementation. Single-visit endodontic treatment of maxillary left central incisor was completed [Figure –]. In next visit, postspace was prepared using peeso reamers (Mani) till size #3 and 5 mm of apical seal was preserved [], followed by intraradicular impression with addition silicone (President). Finally, impression was poured with die stone to obtain a model, which served as a guide during the making of the post.\nA freshly extracted permanent maxillary canine was collected and thoroughly cleaned to remove soft tissues and then autoclaved at 121C for 15 min.[] Using a diamond disk, tooth was sectioned mesiodistally along the long axis of the tooth and then cementum was removed using tapered fissure diamond bur under coolant to obtain a dentin post [, ]. Model was used as a reference in orienting shape, thickness, and length of the dentin post which was verified radiographically [, ].\nThe internal surface of crown segment was prepared to receive dentin post []. Finally, both biological dentin post and crown were autoclaved just before cementation.\nIn the next visit, flap surgery was planned to expose the fracture line which was subgingival on distopalatal aspect. After administration of local anesthesia (2% lignocaine with 1:80,000 adrenaline), flap was raised to expose the fracture line [], ViscoStat (Ultradent) was used for hemostasis, then retraction cord (#000, Ultradent) was inserted to assist the restorative procedures. The dentin post, inner surface of crown, and the root canal were conditioned with 37% phosphoric acid for 15 s, followed by the rinsing, drying, and application of dual cure-bonding agent (Adhesive [A + B] of Paracore, Coltene) []. Finally Paracore (a dual cure resin cement) was injected into the prepared post space and the dentin post was inserted into the root canal. Simultaneously crown segment was also reattached in place followed by curing for 40s on each surface. [, ]. Then all margins were polished and the flap was repositioned with the sutures and the necessary occlusal adjustments were made. Postsurgical instructions were given to the patient. After 7 days, sutures were removed, and the patient was rescheduled for the next appointment for direct restorations of 11 and 12. In the final visit, direct composite buildup was done using layering technique in 11 and 12 [, ].\nAfter 1-year follow-up, the clinical and radiographic findings [–] showed that the adaptation of reattached crown and biological post as well as the esthetics and the tooth function was preserved. The treatment outcome of this case so far could be considered a success based on the characteristics of clinical and imaging outcomes given by Estrela et al. in 2014. Clinical examination included the absence of tooth pain, absence of periodontal pocket, tooth with retained definitive restoration and in masticatory function, while imaging aspects included the absence of periapical radiolucency.[] |
An 8-year-old male presented to the emergency room with a 2-day history of shortness of breath and wheezing. He had a history of moderate-persistent asthma and had several emergency department visits over the past 2 years for similar complaints attributed to asthma exacerbation. He was on albuterol and inhaled corticosteroids for his persistent asthma, but he reported that it was not effective in relieving his exacerbation. He was admitted for acute asthma management due to increasing work of breathing and wheeze. He denied any dysphagia or odynophagia.\nThe patient’s growth was appropriate, respiratory rate at 40 breaths per minute, heart rate at 130 beats per minute, blood pressure of 90/60 mm Hg, and pulse oximetry of 95% in room air. The rest of the physical examination was unremarkable.\nOn admission, his frontal and lateral chest X-rays showed only an incidental finding of right tracheal deviation. His previous chest X-rays showed persistent right tracheal deviation, which was not of prior concern due to its mild nature. A transthoracic echocardiogram revealed mild tracheal compression of the left atrium just above the bifurcation, at the level of descending aorta, either by the aorta or the surrounding structures. A contrast-enhanced computed tomography (CT) scan of the chest demonstrated a right-sided aortic arch with aberrant left subclavian artery and KD, mild tracheal deviation, and moderate esophageal deviation and compression ( and ). Pulmonary function tests read mixed an obstructive/restrictive pattern, and barium swallow demonstrated posterior narrowing of the proximal thoracic esophagus. Esophageal compression was likely secondary to right-sided aortic arch with aberrant left subclavian artery sling. With these findings on imaging, foreign body aspiration, tracheomalacia, retropharyngeal abscess, cysts, neoplasms, and bony dysplasia were subsequently ruled out.\nOur patient’s respiratory distress improved with albuterol and systemic steroids. We discharged him with an increase in his albuterol and inhaled corticosteroid doses and a course of prednisolone. He had an outpatient follow-up for surgical intervention (left thoracotomy division of the vascular ring) of his KD. |
We present the history of an 82-year-old man with chronic lymphatic leukemia (B-CLL), diagnosed in 2004 who received rituximab, fludarabine and cyclophosphamide for four cycles between May 2010 and October 2010 as first-line therapy. The past medical history showed arterial hypertension and prostate cancer treated with radical prostatectomy in 2005.\nIn July 2014, an SCC of the skin located in the scalp was diagnosed and completely resected without any signs of metastases. At that time, there was no sign of progressive B-CLL. In January 2015, the patient presented with progressive lymphadenopathy, night sweats, fever and weight loss and a worsening of peripheral blood count with a decrease in hemoglobin (from 11 to 9 g/dl) and a reduction of thrombocytes (from 60 to 20 × 109/l). Further examinations revealed a progression of the B-CLL with generalized lymphadenopathy and an increase in monoclonal B-CLL population (up to 65%) in peripheral blood detected by FACS. Therefore, salvage therapy with ibrutinib was started in January 2015.\nAfter an initially very good response to ibrutinib with fast significant lymph node regression and improvement of the peripheral blood count, in February 2015, he presented with progressive painful cervical swelling and numbness of the right side of his face. At that time, the B-CLL was in stable remission, and therapy with ibrutinib was stopped immediately. An MRI scan revealed a progressive cervical tumor. A lymph node biopsy showed an infiltration of a keratinizing, moderately differentiated squamous cell cancer of the skin due to a metastatic manifestation of the squamous cell cancer of the scalp diagnosed in July 2014. In February, first-line treatment with primary radiochemotherapy (cisplatin 20 mg/m2 days 1–5 and radiation with 60 Gy) was initiated. He received a total of three cycles until April 2015. There were no signs of metastases at the end of therapy. However, the primary tumor, located retroauricular, was not resectable. In August 2015, a CT scan showed progressive disease with new tumor manifestations in the lung, cervical lymph nodes and mediastinum. Subsequently, in August 2015, a second-line chemotherapy consisting of cetuximab and docetaxel was initiated.\nIn September 2015, a CT scan revealed a refractory disease with significant tumor progression after two cycles of chemotherapy. Further analysis detected positive PD-1 expression of the tumor (fig. ). In accordance with the patient's insurance, a therapy with nivolumab was started. From October 2015 until January 2016, nivolumab was administered at a dose of 3 mg/kg intravenously every 2 weeks. Therapy was well tolerated and side effects were unremarkable except for generalized muscle weakness and fatigue. In particular, there were no severe infections or signs of autoimmune disease. Routine follow-up laboratory parameters remained mostly normal. During nivolumab therapy, no changes in immune cells were detected with a reduced, but stable T-cell count (CD4+ and CD8+ <200/μl) before, during and after treatment with nivolumab.\nAfter four applications of nivolumab, a CT scan was performed that demonstrated a significant response with marked size regression of the main cervical tumor manifestation and of the pulmonal manifestations. Abdominal lymph nodes, probably due to the B-CLL showed stable disease. Therefore, therapy with nivolumab was continued and was well tolerated. The initial weakness improved after the third administration. In February 2016, after eight administrations of nivolumab, the patient presented with a rapid progressive decrease of hemoglobin. At that point, there was no sign of progressive CLL as lymph node manifestations of CLL were stable with a constant proportion of monoclonal B-lymphocytes due to B-CLL in peripheral blood detected in FACS analysis.\nFurther laboratory examinations confirmed the diagnosis of acute hemolysis with detection of irregular antibodies. Detailed diagnostics detected warm antibodies with positive direct agglutinin test of anti-IgG and anti-C3.\nConsequently, nivolumab was terminated and treatment with prednisolone 80 mg/day was started. After 2 weeks, hemolysis was stopped and peripheral blood count stabilized. At re-evaluation in February 2016, after a total of eight administrations of nivolumab, CT imaging showed a good response with further regression of the cervical, thoracal and mediastinal tumor manifestations (fig. ). Due to the observed hemolysis, a progressive splenomegaly was diagnosed. One month after termination of nivolumab treatment, the patient presented in good physical condition with recovered peripheral blood count and no signs of hemolysis or tumor progression. Finally under third-line therapy with nivolumab, squamous cell skin cancer showed a satisfying response for a duration of at least 5 months (from October 2015 until March 2016). |
A 43-year-old Chinese Han man was admitted to our hospital because of edema of lower limbs bilaterally for 1 year, exacerbating with gross hematuria in the last 2 months.\nOne year ago, he developed pitting edema of lower limbs and also found bubbles in urine at the same time. He visited the local hospital and the diagnosis of lower limbs varicose vein was made. Ten months later, his edema aggravated consciously and he presented with the whole course gross hematuria. The routine urinalysis showed proteinuria (3+) and hematuria (3+). Urinary protein excretion amount was 3.7 g/24h. The serum creatinine value was in the normal range and increased to 2.47 mg/dL one month later.\nThe past history revealed that he was a coal miner for 30 years and diagnosed as silicosis 3 months ago. He presented with hypertension for 4 years and it could be controlled at the range of 120–130/80–90 mmHg by regular medications. He did not abuse alcohol, cigarettes, or other drugs.\nAfter admission, physical examination revealed that his temperature was 36.5 °C, respiratory rate was 20 breaths/min, pulse rate was 76 beats/min and blood pressure was 130/80 mmHg. There was no jaundice, rash and bleeding by skin examination and the superficial lymph nodes was not touched. Pitting edema of lower limbs was found bilaterally.\nsummarized all the laboratory indices after his admission.\nHigh-resolution computed tomography (HRCT) showed that there were multiple small nodular lesions on both lung fields and multiple calcifications were on the left upper lobe.\nUltrasound showed that the left and right kidneys were both in normal size. No stenosis or thrombus of renal artery and vein were found by Doppler ultrasound.\nThe patients presented with nephritic syndrome and acute kidney injury (AKI), which could not be excluded with silicosis associated renal disease. Thus, renal biopsy was crucial for the diagnosis and it was performed after his admission.\nHis renal biopsy specimen was examined by light microscopy, immunofluorescence, and electron microscopy. By light microscopy, 23 glomeruli were included in the specimen. One glomerulus was ischemic sclerosed and the remaining glomeruli manifested as mild mesangial cell and matrix proliferation with segmental endocapillary hypercellularity. Fuchsinophilic deposits were observed in mesangium. There were one cellular crescent and four fibro-cellular crescents. Tubular epithelial cells showed cytoplasmic vacuolization and focal loss of brush border with focal tubular atrophy. There was moderately interstitial infiltration of lymphocytes, mononuclear cells and a few eosinophils with focal interstitial fibrosis. Arterioles were thickened with hyalinosis. Immunofluorescence revealed lump and granular staining of IgA (3+) and C3 (3+) in mesangium and others including IgG, IgM, C1q, and fibrin were all negative ().\nBy electron microscopy, mild mesangial expansion with electron dense deposits in mesangial and para-mesangial matrix were observed. No remarkable changes were seen in glomerular basement membrane and the foot processes of podocytes were effaced diffusely ().\nThe final pathological diagnosis was focal proliferative IgA nephropathy (Oxford classification: M1E1S0T1) and acute tubulo-interstitial nephritis.\nBased on the renal pathological findings and his occupation, we proposed that the kidney disease might be associated with the silica exposure. Then, the kidney sections of the patients were further scanned using polarization microscopy for the quantity of silica or silicon dioxide crystal deposition. However, we did not find silica or silicon dioxide crystal deposition neither in glomerulus nor tubulo-interstitial areas ().\nAs the NLRP3-mediated inflammation might be involved in the oxalate nephropathy and silicosis, the MARCO, NLRP3, Caspase-1, ASC, IL-1β, and IL-18 were further stained by immunohistochemistry which were all positive in both glomerular and tubulo-interstitial areas in our patient and they were all virtually negative in the normal control (normal part of one nephrectomized kidney due to renal carcinoma) (). Furthermore, we selected one primary IgA nephropathy patient (Oxford classification: M1E1S0T1) as the disease control and we found that the expressions of MARCO, ASC, Caspase-1, and IL-1β were similar with our patient. However, the staining of NLRP3 was significantly higher in tubulo-interstitial areas than that in glomerular areas in the disease control, and the staining of IL-18 was specifically expressed in the distal convoluted tubules and some part of glomerular areas in our patient and it was dispersive around glomerular and tubulo-interstitial areas in the primary IgA nephropathy patient ().\nAs the patient was diagnosed as IgA nephropathy combined with acute tubulo-interstitial nephritis, the prednisone (30 mg/d) was then initiated with tapering regularly in combination with ACEI treatment. The patient got a significant improvement both for renal function and proteinuria after 2 months, which kept stable still now. He also changed his job to remove the occupational factor. His laboratory data at follow-up were showed in . |
A 45-years-old male engineer with no history of trauma, presented to us with progressive upper back pain for two months that eventually became severe and disabling. The pain was non mechanical and was present even at rest and night. He denied any constitutional symptoms. He had no significant medical history except that he was a smoker for over 20 years.\nOn neurological examination, patient indicated altered sensation from T8 dermatome and below; however, upper and lower limb motor power was normal. He had an unsteady gait and exaggerated deep tendon reflexes in both lower limbs.\nInitial X-rays of the thoracic spine were unremarkable, but MRI demonstrated abnormal marrow replacement and enhancement of the entire T6 vertebra including its posterior elements and right 6th posterior rib. MRI axial cuts at T6 level revealed right postero-lateral epidural extension of the lesion causing severe spinal canal, right lateral recess and right exit foraminal stenosis (, ). In addition, an irregular right lung nodule was noted. CT evaluation confirmed the vertebral involvement () and the presence of a lobulated right lung nodule with emphysematous changes in bilateral upper lobes. Further radiological assessment revealed no other lesion elsewhere.\nDuring the course of the work up, he developed bilateral lower limb weakness and was unable to stand. A diagnosis of thoracic myelopathy due to cord compression was made and immediate surgical management in the form of T6 decompression laminectomy along with T3-T8 posterior instrumentation was performed (). Surgery was uneventful; patient had good recovery and was ambulating independently.\nTissue biopsy from T6 right pedicle revealed presence of metastatic tumour cells with moderate amount of eosinophilic cytoplasm in the marrow spaces suggestive of a metastatic carcinoma with neuro-endocrine features (). Tumour cells were positive for cytokeratin AE1/3, carcinoembrionic antigen (CEA), chromogranin and synaptophysin. Proliferation marker Ki67 was found to be 40%.\nEarly post-operative MRI showed adequate decompression of the spinal canal at T6 level and the patient had interval resolution of symptoms; but there was abnormal fluid collection from the surgical site extending up to the subcutaneous layer, likely to be seroma, which was conservatively managed. Chemotherapy with Carboplatin and Etoposide was initiated after satisfactory wound healing three weeks after surgery.\nOne month after the index surgery, patient developed significant motor deficit in the lower limbs (Right L2-L5 = 2/5; Right S1 = 4/5; Left L2-S1 = 4/5) and had bowel and bladder incontinence. An urgent MRI was done which revealed recurrence of the lesion causing near total obliteration of the spinal canal ().\nA second surgery at this stage was considered high risk and the decision was made to initiate concurrent site specific T4-T7 radiotherapy (30 Gy in 10 fractions) along with chemotherapy. Interestingly at the end of radiotherapy and four courses of chemotherapy (4 months post-op), there was significant clinical improvement of neurology and complete resolution of the soft tissue enhancement surrounding the spinal cord was evident in the MRI (). With appropriate physiotherapy and rehabilitation, patient gradually regained full power in both lower limbs by 6 months. He continues to be under oncology follow up and is ambulant with support. |
A 90-year-old man was brought to the emergency room due to acute onset of severe generalized weakness, sudden onset of dyspnea, pleasure whistling in respiration, acute dysphagia to both liquids and specially solids and inability to speak, from 2 days ago. He had one time vomiting at onset of the symptoms. He had medical history of achalasia, recurrent pneumonia in last months and head trauma 2 months ago with sub-arachnoids hemorrhage and several convulsions. On arrival to hospital, his vital sign were as oral temperature: 37.8°C, heart rate: 100/minute, respiratory rate: 16/minute, blood pressure: 100/80 mmHg with 95% oxygen saturation on room air. He was completely alert but could not talk. In physical exam except for severe cachexia and superficial ulcers on two forearms, there was no other finding. Lung exam was incomplete due to severe illness and superficial respiration. He did not have stridor and wheezing! None of examiners noticed to his respiration with pleasure whistling and the history of losing his upper dental prosthesis from two days ago. Posterior-anterior chest x-ray revealed bilateral consolidations with the most prominence in the right middle lobe (). Chest computed tomography (CT) scan revealed the same finding as well as bilateral pleural effusion (). He was admitted to the Infectious Disease ward with diagnosis of aspiration pneumonia. In next day, his dyspnea guts worst so made the corresponding physician to take a lateral neck radiography (). A large foreign body was lodged above epiglottis between oropharynx and hypopharynx. On physical examination, his lost upper complete upper denture with 5 x 5 centimeter size was seen in his pharynx behind the tongue and was manually extracted with fingers. His three days dyspnea, whistling, inability to talk and dysphagia was dramatically improved. He was treated for aspiration pneumonia due to last convulsions and was discharged in a stable condition after one week. In third visit after two months, he had no alimentary and respiratory sign and symptoms with good condition. |
A 20-year-old woman with a giant convexity meningioma was introduced to our hospital. At the age of 17 years, the meningioma was incidentally noted after examination of the left lower limb. She visited a local hospital because of slow aggravation of chronic headache, vomiting, and somnolence. Consciousness improved after the administration of steroids and glycerol, and she was transferred to our department for surgery. The frontal calvaria were irregularly thickened on computed tomography. Gd-enhanced MRI on admission revealed a giant homogeneous enhanced tumor within the frontal calvaria associated with extracranial and intradural extensions []. The superior sagittal sinus (SSS) was invaded but not completely occluded. Collateral circulation developed through bridging veins around the tumor []. To reduce blood loss, we decided to preserve the tumor around the SSS. Hyper-osteosis from the intraosseous lesion was considered compressive and symptomatic because high intracranial pressure may cause neurological symptoms.\nStaged surgical resection and cranioplasty were planned. The skin flap was turned forward after a coronal skin incision, and the subcutaneous tumor lesion was dissected from the serial bony lesion [Figure and ]. Cutting into some pieces of the cranium was difficult because of marked thickness; therefore, burr holes were placed in normal cranium, and we divided the abnormal cranium and subdural lesion. During this procedure, approximately 2.1 L of blood was lost from the SSS, necessitating blood transfusion. The origin of the tumor was the parasagittal intraosseous layer and the tumor extended intra-durally with localized occlusion of the SSS [Figure and ]. Pial tumor invasion was also observed in the cortical surface. A large amount of tumor tissue was dissected from the cortical surface and the dural-extending lesion was resected. Dissecting the hyper-osteotic skull from the dura matter was difficult because the working angle for the dissecting plane was limited and because of serial invasion from the intraosseous to cortical surface. The frontal cortical surface was damaged. To preserve the SSS, the tumor removed was classified as Simpson grade 3 []. Hydrocephalus and subcutaneous cerebrospinal fluid (CSF) collection developed as complications and were managed under external drainage. A lumbar abdominal shunt was placed and subcutaneous CSF collection improved. On pathology, intertrabecular spaces were infiltrated by the meningothelial meningioma, and the intracranial tumor lesion had the same features and contained trabecular tissue. MIB-1 staining was negative in the intracranial tumor and extracranial tumor (1% and 2%–3%, respectively). The patient was transferred for rehabilitation. Seven months after tumor resection, rehabilitation was completed and joint cranioplasty with plastic surgery was planned. Due to the extensive skull defect, we decided to use a titanium plate []. The temporal posterior periosteal flap, which was continuous from the bilateral temporal periosteal flap, was lifted and sutured to lie below the skin incision such that the titanium plate was not directly exposed. The surgery was completed after the placement of a subcutaneous drain [Figure and ]. No recurrence was noted 1 year after the surgery. We've obtained her consent for this report |
A 45-year-old woman developed ocular pain and blurred vision in her right eye a month prior to presentation. She initially visited a separate clinic and received intravenous injections of antibiotics. These proved ineffectual, and she was referred to another hospital, where she was diagnosed with iritis with ciliary injection, keratic precipitate, hypopyon, and posterior synechia in her right eye. Topical betamethasone and oral prednisolone (30 mg/day) were applied. Although this treatment proved effective in healing the iritis, the redness and pain in her right eye deteriorated and she was referred to our hospital. Her left eye had mild visual impairment due to a childhood trauma. She did not have any medical history of systemic disease.\nThe initial ophthalmic examination disclosed a best-corrected visual acuity (BCVA) of 20/20 in the right eye and 20/32 in the left eye. The intraocular pressure was 15 mm Hg in the right eye and 18 mm Hg in the left eye. A slit-lamp examination demonstrated no sign of uveitis or retinal disease besides conjunctival injection and scleral venous plexus congestion in the right eye. Both eyes showed substantial anterior chamber depth without peripheral anterior synechia. The clinical course is described in figure . Results of the laboratory investigations of serum including antinuclear antibodies, anti-double-stranded DNA antibodies, rheumatoid factors, and antineutrophil cytoplasmic antibodies were unremarkable. Serological analysis indicated that there was no active infection.\nThe patient continued to receive topical betamethasone and oral prednisolone (10 mg/day), and subconjunctival triamcinolone injections were additionally administered, resulting in modest improvement. Neither topical cyclosporine nor topical antibiotics showed any effect on her ocular symptoms. One month later, brain magnetic resonance imaging indicated that there was no thickening of the posterior sclera or other abnormalities. When oral prednisolone was tapered, the patient's scleral congestion and ocular pain were aggravated. After admission, two sets of pulse therapy with intravenous methylprednisolone (1 g/day for 3 days) following oral prednisolone (40 mg/day) were applied, yielding a moderately positive response. She then received systemic cyclosporine (3 mg/kg/day), administered orally to treat aggravated scleritis, and her ocular symptoms subsequently declined. When the oral prednisolone treatment was terminated and the cyclosporine dosage was reduced to 2 mg/kg/day, the scleritis relapsed. When cyclosporine was increased to 3 mg/kg/day, the trough value was 37 ng/ml, and her right eye developed peripheral cornea edema (fig. ). Upon increasing the amount of cyclosporine to 4 mg/kg/day, her ocular symptoms improved (fig. ). Although there was another relapse 3 months later, the cyclosporine dosage was retained. Since then, no relapse has occurred even after tapering the cyclosporine. During the clinical course, there was a temporal intraocular pressure elevation which could be maintained by topical antiglaucoma medication. Blood tests were examined monthly to check the trough value of cyclosporine and to monitor the cell counts and biochemical markers, particularly the renal function marker. No side effects resulting from systemic cyclosporine administration were observed. |
A 40-year-old male not known to have any chronic medical illness, presented complaining of epigastric and left upper quadrant pain for 1 month, associated with intermittent nausea and vomiting, and aggravated by fatty meals, with no other associated symptoms. He had frequent visits to the emergency department where he was managed with analgesia and antacids with mild symptomatic improvement. Clinical examination was unremarkable with no evidence of jaundice or abdominal tenderness. His blood test results showed a normal complete blood count, kidney function, and liver function. Chest X Ray revealed dextrocardia with stomach fundic gas shadow on Right side (). Abdominal ultrasonography revealed transpositioning of the solid organs with a left sided liver and gallbladder with a solitary stone and mild wall thickening. We elected to perform a Magnetic Resonance Cholangiopancreatography to delineate the anatomy and to rule out any anomalies within the biliary tree. It confirmed the previously noted findings, showed no evident anomaly within the biliary tree, and confirmed the diagnosis of situs inversus totalis (, ). The patient was Scheduled for an elective laparoscopic cholecystectomy.\nThe Operating room equipment arrangement was adjusted as Mirror Image of Routine Laparoscopic cholecystectomy (). The Monitor was placed on left side of the patient. The surgeon with the camera assistant were on right side of the patient and the first assistant was on left side of the patient. The abdomen was scrubbed and draped in the standard aseptic technique. The first infraumbilical 11 mm trocar introduced and pneumoperitoneum induced using the open technique. Three 5 mm trocars were placed, at the xiphisternum which was used for the surgeon’s left hand, at the left midclavicular line 2 cm below the costal margin which was used as working port for the surgeon’s right hand and at left anterior axillary line 5 cm from the costal margin which was used for retraction of the gallbladder fundus by the second assistant, respectively. Inspection of the abdominal cavity confirmed the presence of situs inversus totalis, with the liver and the gallbladder positioned in the left side (). The Calot’s triangle was identified. The peritoneum overlying the gallbladder infundibulum was then incised and the cystic duct and cystic artery identified and circumferentially dissected, till the critical view was obtained. The cystic duct and cystic artery were then doubly clipped and divided, through the subcostal port using the right hand. The gallbladder was dissected from its peritoneal attachments using electrocautery and was retrieved using Endoscopic bag through the infraumbilical port. The total operative duration was 80 min, which was longer than the conventional laparoscopic cholecystectomy performed in patient without underlying anatomical variation. It can be attributed to the modification in the technique required to adjust to the mirror image anatomy.\nThe patient had an uneventful postoperative course and was discharged on postoperative day 1. Pathological examination of the gallbladder confirmed the presence of gallstones with chronic cholecystitis. No postoperative complications were noted during his follow up in the outpatient department. |
A 44-year-old Chinese female with left invasive ductal breast cancer underwent a modified radical mastectomy in April 2005. The pathological stage of her cancer was T2N0M0 with intermediate grade and lymphovascular invasion. The primary tumor was ER, PR, and HER2 positive and phosphatase and tensin homologue (PTEN) negative as determined by immunohistochemistry (IHC). The expression of ER and PR was scored according to the Allred score. HER2 status was scored as positive if >30% of tumor cells showed strong (3+) membrane staining, and PTEN status was designated as positive if tumor cells showed positive staining by IHC. The patient was treated with CAF (cyclophosphamide, adriamycin, and fluorouracil) adjuvant chemotherapy for six cycles and tamoxifen for 2 years without radiotherapy or trastuzumab. Metastases to the supraclavicular and cervical lymph nodes and left chest wall relapse were found in December 2007. Thus, the disease-free survival was 32 months.\nFrom December 2007 to August 2012, the patient underwent multiple-line rescue treatments including several cytotoxic agents, HER2-targeted therapies, and endocrine therapies used for breast cancer (). Pretreatment biopsy and pathology results were not available. The rescue treatment was started with chemotherapy, but this was switched to endocrine drugs due to the adverse effects of chemotherapy. The patient obtained clinical benefit from endocrine therapy. The recurrent tumor in the left supraclavicular lymph nodes was HR negative by IHC and HER2 positive by fluorescence in situ hybridization detection in two hospitals in May 2009. Then, chemotherapy and HER2-directed therapy as main choices were applied, and endocrine therapy was also used due to the intolerance or lack of response to chemotherapy. Among these regimens, two regimens provided clinical benefit, namely, anastrozole combined with goserelin for 11 months and exemestane plus lapatinib for 7 months during the earlier treatment. In contrast, a total of seven regimens containing trastuzumab and two regimens containing lapatinib all failed ().\nAfter discussion of various therapeutic options including palliative care, in 2012, we decided to treat the patient with everolimus (5 mg/d orally) in combination with intramuscular fulvestrant (500 mg once/28 days). The response and side effects of the regimen are shown in . After 6 days, we increased the dose of everolimus to 10 mg/d for 34 days., Measurable lung lesions diminished modestly as observed by computed tomography (CT) examination (). The changes in target lesions based on the maximum reduction of the sum of lesion diameters are shown in . Side effects included third-degree stomatitis and liver toxicity and second-degree hematologic toxicity. After discontinuation of everolimus for 10 days, the side effects were relieved and eventually disappeared. When the patient took 5 mg everolimus daily, the lung lesions increased slightly. We therefore increased the dose to 5 and 10 mg/d alternately, with an estimated daily dose of 7.5 mg, and the lesions diminished again. Subsequently, everolimus was reduced to 5 mg daily or treatment was discontinued due to fatigue and other adverse events. Treatment was resumed again at 5 and 10 mg/d alternatively and after approximately six months of treatment, the patient could tolerate the full dose (10 mg/d) of everolimus.\nAt the first appearance of tumor progression detected by CT on June 7, 2013, the patient’s pleural effusion was extracted for pathologic examination and molecular profile testing (performed by Caris Life Sciences, Irving, TX, USA). The results confirmed that the tumor was a HR-negative, HER2-positive adenocarcinoma that contained a PIK3CA gene mutation and was positive for PTEN expression (). The patient continued everolimus treatment until disease progression was confirmed on June 24, 2013. The response was stable disease as evaluated by Response evaluation criteria in solid tumors, and progression-free survival (PFS) was 10 months. Thereafter, the patient was treated with trastuzumab, emtansine, and sorafenib, individually, with no measurable responses. The patient died on October 27, 2013, with overall survival time of 102 months. Written informed consent was obtained from the patient for publication of this Case Report and any accompanying images. This case report was approved by the Ethics Committee of Affiliated Hospital of Academy of Military Medical Sciences. |
A 27 year-old man visited our outpatient clinic in the Department of Psychiatry, Hanyang University Guri Hospital in Gyeonggi Province, Korea, complaining of fear of using public restrooms and worrying about others watching him urinate. This fear had begun 10 years earlier when he was a sophomore in high school; since then, he had persistently avoided such places, which made it difficult for him to travel or engage in social activities. He managed to enter and use the restroom if only one or two others were in it, but could not use a restroom with several other people in it. This condition had greatly impaired his academic progress and interpersonal activities. During the course of his illness, he had attended a psychiatric clinic and received a prescription for antidepressants and antianxiety medications, but there had been no improvement over one year of use. In addition, he had attended six sessions of individual cognitive behavioral therapy for social phobia but had terminated the treatment prematurely due to a lack of perceived improvement. He had heard out about EMDR from the media and visited our clinic with the hope receiving the treatment.\nThe patient met the DSM-5 criteria for social anxiety disorder in that he had consistently experienced marked fear and anxiety about using public restrooms while others were present, and he feared that other people would notice his anxiety and embarrassment.\nHe was referred to EMDR therapy with the first author (HP) and privoded informed consent for receiving DeTUR and participating in a case report. One week before initiation of the DeTUR, his baseline symptom level was measured with the State Anxiety Inventory (SAI), the Social Avoidance and Distress Scale (SAD), and the Fear of Negative Evaluation (FNE). He completed the questionnaires again one week after the final session and at the one-year follow-up. The patient attended eight weekly 60-minute sessions, at the end of which he had no anxiety about using any public restroom, and his self-reported symptoms on the three measures had reached nonclinical levels. State anxiety (score range 20-80, cut-off 52) decreased from 56 pretreatment to 39 post-treatment and 26 after one year. Social avoidance and distress (range 28-140, cut-off 61) decreased from 98 pretreatment to 53 post-treatment and 44 after one year. Fear of negative evaluation decreased from 48 pretreatment to 21 post-treatment and 26 after one year (range 12-60, cut-off 42).\nIn the first session, the patient was given information about EMDR therapy, a brief history was taken, and he was taught about a safe place exercise: guided imagery for controlling possible distress between sessions. The therapist encouraged him to use this image when he experienced distress outside the sessions. His second session began the actual DeTUR protocol, and the hierarchy of triggers to avoid was established after discussion with the therapist (). From session 3 to 8, each of these eight triggers was desensitized until the LOU decreased to zero, starting with T1 (the lowest LOU) and ending with T8 (the highest). As the sessions progressed, the patient gradually gained control of his fear and anxiety about public urination. After the fifth session, he voluntarily exposed himself to a situation addressed in the previous session, although he had not been given any suggestions or homework about in vivo exposure. At the final session, he reported that he was able to visit and use any public restroom. |
We present the case of a 42-year-old man who presented to the emergency department with a complaint of abdominal pain and diarrhea for 3 days. The abdominal pain started in the periumbilical region and was shifted to the right lower quadrant of the abdomen. The pain started gradually and had been progressing in severity. He described the pain as a stabbing in nature. It was exacerbated by movement and food intake. The pain was partially relieved by oral analgesic medications like paracetamol. The pain was associated with low-grade fever and decreased appetite. The patient also complained of diarrhea with five bowel motions/day. The stools were watery with no mucus or blood. He reported that diarrhea developed after he received an oral antibiotic therapy (cefuroxime) for a recent upper respiratory tract infection.\nThe past medical history of the patient was remarkable for diabetes mellitus that was well-controlled with oral antidiabetic agents. He did not undergo any previous abdominal surgeries. He had a smoking history of 15 pack-years. He had never drunk alcohol before. He worked as a taxi driver. The family history was unremarkable for any inherited gastrointestinal disorders.\nUpon examination, the patient appeared sick. He was not pale, jaundiced, or cyanosed. Vital signs revealed tachycardia (115 bpm), low-grade fever (37.5℃), normal respiratory rate (14 bpm), and maintained blood pressure (122/80 mmHg). The oxygen saturation was 99% on room air. Abdominal examination revealed a soft abdomen with diffuse tenderness. However, the tenderness was more pronounced in the right iliac fossa with a positive rebound sign. Further, the Rovsing sign was positive. Initial laboratory investigation revealed elevated leukocyte count and elevated inflammatory markers, including erythrocyte sedimentation rate and C-reactive protein. The renal and hepatic profiles were within the normal limits (Table ).\nIn light of the aforementioned clinical information, the patient was diagnosed as having acute appendicitis. A CT scan with intravenous contrast was performed to confirm the diagnosis. The scan demonstrated colonic wall thickening with edematous haustral folds suggestive of pseudomembranous colitis. Further, an endoluminal lesion was observed in the cecum with an average size of 6 cm. The lesion was well-defined and had a homogenous fat density with no solid component. The mass was causing a partial colonic obstruction. Such findings conferred the diagnosis of cecal lipoma (Figure ).\nThe patient was prepared for an emergency laparoscopy for further evaluation and management. The operation was done under general anesthesia and the patient was placed in the supine position. Limited segmental rection of the cecum with appendectomy was performed. The estimated blood loss was 10 mL and the total operative time was 100 minutes. The patient tolerated the operation with no complications. He had an uneventful recovery. Histopathological examination of the resected sample revealed the diagnosis of cecal lipoma and the associated acute appendicitis. The patient was discharged on the fifth postoperative day. After 3 months of follow-up, the patient remained asymptomatic with no active issues. |
A 43-year-old man with a past medical history of gout presented to our hospital with one-month history of progressive lower extremity weakness, gait instability, and acute back pain. The patient reported he was subjectively diagnosed with viral meningitis one month prior to presentation to the emergency department (ED) with symptoms at the time including cough, fever, anorexia, malaise, fatigue, myalgias, cervicalgia/neck stiffness with flexion and extension, mild photophobia, headache and two-week history of scaly erythematous macular rash on his proximal medial upper and lower extremities. The patient refused to undergo lumbar puncture at the initial onset of his symptoms and thus a diagnosis of any infectious intracerebral/intrathecal process was never confirmed. At the onset of the patient’s symptoms, he had tried over-the-counter analgesics with some relief of his headaches though his generalized pain persisted. He initially underwent extensive laboratory studies at the onset of his symptoms ordered by his primary care physician one month prior to his presentation including rheumatologic evaluation and screening tests for tick-borne infections including Lyme serologies, however they were unremarkable, except for mildly elevated AST 79 and ALT 79, elevated CRP 4.95 mg/dL, and complement C3 227 mg/dL. His symptoms persisted and changed requiring hospital evaluation.\nAt presentation to our hospital, the patient reported progressive weakness and severe radicular lancinating pain going from his lower back to his heels worse on the right side that is worsened with sitting and supine, emotional lability along with depression and anxiety. He had also noticed occasional action tremor in hands interfering with fine motor tasks, and mentioned feeling tremor in his legs causing imbalance and instability though with no falls. He denied any bowel and bladder dysfunction, although he reported an episode of premature ejaculation a couple weeks prior to presentation.\nHis neurological exam was normal including strength, sensory, and reflex testing except for an unsteady wide based gait. We obtained a magnetic resonance imaging of lumbar spine with and without contrast that showed slightly thickened enhancement along the surface of the conus medullaris as well as enhancement of the nerve roots of the cauda equina, pronounced degenerative disc disease at L4-L5 with a broad-based disc-osteophyte complex, and mild bilateral facet arthropathy at L4-L5 results in mild-moderate bilateral neural foraminal stenosis as shown in . The nerve conduction studies and electromyography of his bilateral lower extremities were normal. A lumbar puncture was performed and the patient’s cerebrospinal fluid (CSF) analysis showed lymphocytic pleocytosis with white blood cell count of 225 and elevated protein of 77 and decreased glucose 38. Ultimately his serum LD Western Blot came back reactive with three IgG proteins and two IgM proteins, and one LD IgM band in CSF also came back positive.\nThe patient was started on oral doxycycline 100mg BID for 5 days prior to placement of a PICC line for starting the patient on IV ceftriaxone 2g daily for 4 weeks in treatment of Lyme radiculitis and meningitis. On follow up in clinic two and a half weeks after starting medical therapy the patient noted his symptoms were significantly improved including resolution of the pain, weakness, constitutional and affective symptoms, while he still had some ambulatory difficulties. |
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