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A 14-year-old boy was admitted to hospital with hematuria. Results of complete blood count and biochemical test were within normal range. There was hematuria in urinalysis, but urine culture evaluation was sterile. There was no abnormal finding in his history. On abdominal sonography, a lobulated, hypoechoic mass (40 × 28 mm in size) at posterolateral wall of bladder extending to lumen was observed. On Doppler sonography, vascularization was observed at the area of mass. On CT scan, a suspicious lesion (3.5 × 3 cm in size) was observed at left posterolateral wall of bladder (). No enlargement in lymph nodes or finding favoring metastasis was observed in the pelvic region and abdomen.\nOn cystoscopy, a vegetative mass (approximately 5 × 6 cm) that protruded into bladder through a stalk () was observed at left orifice of bladder. In the same session, ureterorenoscopic assessment was performed which revealed that the stalk extended to middle segment of ureter. However, it was failed to observe where the stalk arises at ureter. Open surgery was performed via left Gibson incision in the same session. It was seen that the stalk of polyp originated from the level of iliac bifurcation (). After exposure of dilated region of middle ureter, ureter was opened at superior to polyp and part of ureter harboring the stalk of polyp was excised in segmental manner by preserving the distal ureter as possible (). As frozen sections were reported as fibroepithelial polyp, the mass and its stalk were removed through bladder (). Then, ureteral end-to-end anastomosis was performed. The part of ureter harboring the stalk of polyp was approximately 10 mm in size. Specimen was a reddish-beige tissue (8 × 4 × 1 cm in size) containing papillary projections on surface (as biggest being 2.5 × 2 cm in size) (). Histopathological diagnosis was reported as fibroepithelial polyp. The patient was discharged on day 7 without complication. After 4 weeks, double J stent was removed. No recurrence was observed at 6-months follow-up. |
A 63-year-old healthy male patient underwent a laparoscopic bilateral totally extra peritoneal inguinal hernia repair with two polypropylene meshes in 2010. His medical history reveals no use of any medication, nor diabetes, cardiopulmonary, or other diseases. He had stopped smoking 20 years ago and has a normal body mass index. The surgery went well and according to Dutch practice no tacks were placed. His postoperative recovery was uncomplicated; there were no signs of seroma, hematoma, or recurrence. After almost three years, the patient returned with complaints of swelling in the left inguinal area and lower urinary tract symptoms. The urinary problems were initially successfully treated with an alpha-1-antagonist. Ultrasound and computer tomography (CT) showed a cyst-like fluid collection, approximately 6 cm in diameter. Ultrasound guided aspiration of the cyst was performed due to progressive complaints of restriction of abdominal movement. The cysts' content was a clear serous liquid. Due to persisting urinary problems, a cystoscopy was performed three months later. It showed an abnormal indentation on the ventral side of the bladder, indicative of external bladder compression. CT showed a giant fluid collection of almost 16 centimeters in diameter ventrally of the bladder in Retzius' space, evidently compressing the bladder and causing the urinary problems (). After extensive interdisciplinary deliberation with many experts in Netherlands in the field of inguinal hernia repair, the most minimally invasive approach was chosen: aspiration and sclerotherapy with a tetracycline solution. This time, the cystic content was a brown viscous liquid. Cultures showed no signs of bacterial growth. After initial success, the patient returned with the same symptoms, reappearing after just one week. Ultrasound showed an image of “flying” mesh within a recurrent cystic cavity, which had taken on the original dimensions in less than three weeks. We chose to perform a laparoscopic fenestration due to the localization and the extent of the cyst. Opening of the cyst revealed approximately 600 cc of green-yellow viscous fluid and two floating polypropylene meshes, which could be easily removed by simply pulling them out of the cystic cavity (Figures and ). The cystic cavity was cleaned and roughened. Subsequently, an omental patch was secured with minimal tension in the opening of the cystic cavity, which was left in site (). The fluid and polypropylene mesh were sent for cultures and a piece of the cystic wall was sent for histopathological examination. The patient was discharged the next day and made an excellent recovery. Even though the cystic content appeared similar to pus, cultures showed no growth of bacteria. Histopathology showed that the cystic wall consisted of connective tissue with chronic inflammation without epithelial lining, confirming the diagnosis of a pseudocyst. There was no sign of recurrence of the pseudocyst during the last follow-up, three years after the laparoscopic fenestration. Unfortunately ultrasound showed bilateral recurrence of the inguinal hernia. He chose to refrain from further surgery since the symptoms of the inguinal hernia were not troublesome or problematic in his daily activities. |
A 5-year-old girl presented with proteinuria, serum albumin of 18 g/L, and oedema. She was normotensive with no evidence of significant intravascular depletion. Her renal function was normal. She was commenced on steroid therapy and achieved remission a few days following commencement of steroids, thus confirming steroid sensitive NS. Unfortunately, five months after her steroid treatment was weaned she developed a clinical relapse of her nephrotic syndrome. She recommenced treatment with high dose steroid therapy. Again she responded within one week of commencing steroid treatment. However, when the steroid-weaning regimen was started, she suffered further proteinurea. Despite the return to high dose steroids she failed to achieve remission after 4 weeks of daily oral steroid treatment at 60 mg/m2, and 3 day of IV methylprednisolone at 600 mg/m2. A percutaneous renal biopsy was performed at this point and confirmed minimal change disease. Following the renal biopsy she was commenced on cyclosporine (5 mg/kg/day) therapy and alternate day steroid therapy at 40 mg/m2 and went on to achieve remission in one week. Her renal function remained normal.\nTen months following initial presentation of NS, and three months after commencing cyclosporine treatment, the patient noted an enlarged blind spot in the vision in the right eye. She had no other symptoms suggestive of a raised pressure syndrome or associated systemic illness. At the time of this presentation she was in clinical remission from her NS and was taking prednisolone (1 mg/kg alternate days) and continued on cyclosporine (5 mg/kg/day). Her visual acuity was reduced to finger counting at less than one metre in her right eye with additional total loss of colour vision. In the left eye the visual acuity was 6/9 and colour vision was preserved. Visual field assessment identified an enlarged blind spot with absence of forward vision noted by the patient. Examination revealed bilateral papilloedema.\nMagnetic resonance imaging did not suggest a structural cause for a potential raised pressure syndrome, prompting a LP that confirmed a significantly elevated CSF pressure (> 400 mm H2O). Her intracranial pressure was reduced to 240 mm H2O post CSF drainage at LP and she was then commenced on acetazolamide (30 mg/kg/day). Her cyclosporine dose was reduced to account for the possible contributing role of this drug to increased intracranial pressure []. A computerised tomography venogram study did not identify an acute thrombosis, although a parallel sagittal sinus was noted. On review of her history, she had an episode of severe headaches and vomiting lasting four weeks, two months after her diagnosis of NS. She was completing her weaning dose of steroids at this time and requiring admission to hospital for 2 days. She was conservatively managed. Taken together with the venographic changes, it was clinically plausible that she might have had a sinus venous thrombosis during that event. Her vision was confirmed to be normal at that time, and she did not have any further headaches, particularly throughout her subsequent course of raised pressure syndrome with profound visual failure.\nUnfortunately, her intracranial hypertension remained refractory despite optimised pharmacological therapy with acetazolamide (60 mg/kg/day). With additional and numerous LPs, her vision improved and stabilised with residual deficit. She remained asymptomatic without a headache after the diagnosis of intracranial hypertension was made.\nOne month following diagnosis of intracranial hypertension our patient suffered a clinical relapse of her nephrotic syndrome. At this time she was on a reduced dose of 2.4 mg/kg/day of cyclosporine (trough level of 20 ng/L), which was therefore titrated up to a target trough level of 100 ng/L. Her proteinuria resolved with this management alone and an increase in the dose of steroid therapy was not needed. One week following the increased cyclosporine dose she presented with a further deterioration of her vision with 6/18 vision in the left eye and non-useful vision in the right eye. Acetazolamide dose was increased to 90 mg/kg/d and therapeutic LPs were reinstituted to bring pressures to between 200–250 mmH2O. There was an improvement in the visual acuity in her left eye following CSF drainage. Due to the clinical presentation and reported association of cyclosporine therapy and IIH, her immunomodulation therapy was converted to mycophenolate mofetil (1200 mg/m2/day). As the improvement of her vision was not sustained and her intracranial pressures remained elevated, she underwent an urgent surgical cerebrospinal fluid diversion procedure.\nOver the next few months, her pressures stabilized (acetazolamide 30 – 90 mg/kg/ day was continued and furosemide 1 mg /kg/d was added for a period) in between numerous episodes of raised pressure, presumed to be related to shunt failure. Our patient has undergone two shunt revisions. Three years following initial presentation of her intracranial hypertension she remains severely visually impaired with non-useful vision in her right eye, and 6/36 vision in the left. Her pharmacological treatment for intracranial pressure is currently being withdrawn very gradually. Her nephrotic syndrome remains in remission on treatment with mycophenolate mofetil (600 mg/m2/day) and alternate day prednisolone (20 mg). |
The patient was a 34-year-old woman, G4 L2 Ab1 who had married her cousin 7 years ago. She had a history of two normal vaginal deliveries and one abortion in the 1st trimester. The first pregnancy in the age of 28 had terminated with a normal vaginal delivery (NVD) resulting in a term baby girl who weighed 3150 gr. The second pregnancy had occurred two years later; curettage was done at week 6 due to spontaneous abortion. Her 3rd pregnancy was in the age 32 resulting in a healthy term baby girl weighing 3400gr with NVD. Due to her unwillingness for becoming pregnant she had withdrawal contraception, whereas because of the non-occurrence of menstruation during breastfeeding and 6 months after her last pregnancy, a pregnancy test was requested. Due to the positive pregnancy result, ultrasound study was done which revealed a 10-week spontaneous gestation with 4 gestational sacs and 4 fetuses. There was no case of multiple pregnancies in her or her husband's family.\nShe received prenatal care during her pregnancy but there was no need for prophylactic cerclage. At week 24 of gestation she was hospitalized due to premature contractions. The contractions were controlled with the prescription of pethidine and hydration and she was discharged 3 days later. She was once again admitted at 28 weeks of gestation due to similar contractions; this time she was treated with indomethacin and pethidine and discharged 3 days after the contractions suppression. She also received two doses of betamethasone during hospitalization.\nShe was admitted a week later due to labour contractions. In vaginal examination 2 finger dilatation with no effacement was detected. Serum test results were reported all in the normal range and the vital signs during hospitalization were normal. At this stage she was treated with tocolytics (adalat). The fetuses' health was monitored by Doppler ultrasound imaging, biophysical profile and fetal non stress test (NST). After the labour contractions' suppression and due to the presence of sporadic contractions she was monitored while being hospitalized up to the time of delivery.\nAt 32 weeks and 4 days of gestation, due to the resumption of labour contractions and dilatation progression, after receiving the rescue dose of betamethasone, cesarean section and tubectomy (upon the request of the patient and her husband) was performed. The outcome of cesarean section was 4 fetuses, 3 girls and a boy, quadriamniotic and quadrichorionic. Quadruplet A weighed 1820 gram with an Apgar score of 9 to 10; quadruplet B weighed 1810 gram with an Apgar score of 6-7. Quadruplets C and D weighed 2100 and 1980 gram with an Apgar score of 7-8 and 9-10, respectively. Among the 4 neonates, only quadruplet B was transferred to the NICU; she was discharged after 2 days in good health. and show the quadruplets after birth.\nBecause of atonic uterus during the cesarean section, after the administration of the appropriate dosage of oxytocin and methylergonovine and 800µgr of rectal misoprostol, the uterine arteries were blocked and the B-Lynch suture was done. No blood transfusion was required for the mother and her hemoglobin (Hb) level 6 hours after the operation was 9 g/dl; her pre-operational Hb level was 10g/dl. The mother was discharged 3 days after delivery with no complications.\nFor close follow up, the mother and her newborns were visited two weeks after delivery; they were all healthy and had no problem. The infants were visited once again 6 months later revealing normal physical and mental development in all four. shows the babies at 6 months of age.\nThis project has been approved by Ethical Committee and Vice Chancellor for Research of Mashhad University of Medical Sciences (97/429008). |
A 20 year old Sri Lankan male who was employed as a helper in a grocery, admitted to our unit with weakness of both hands of 1 month’s duration. He was treated for serologically confirmed (Dengue NS1 antigen positive) dengue fever approximately 5 weeks ago at the local hospital and had made an uneventful recovery. He has been given 5 days of inward treatment and the records from the local hospital revealed that he had simple dengue fever with no evidence of fluid leakage.\nFive days after discharge from the hospital he has first noticed the weakness of his right hand when he dropped a glass of water due to poor grip. Weakness was more in the right hand which was his dominant hand and it was slowly progressive over 1 month. At the time of presentation to us he could not write or button on his shirt due the weakness of the hands. Weakness of the left hand was milder than that of the right. The weakness was confined to hands and did not involve forearms or arms. He denied any accompanying numbness, parasthesia or pain.\nOn inquiry he admitted that there was slight weakness of both feet which did not significantly interfere with walking. There was no associated neck/back pain or bladder/bowel incontinence. He did not complain of difficulty in breathing, diplopia, dysphagia, nasal regurgitation, dysarthria or fatigability. He did not give a recent history of trauma to the spine/neck or any preceding diarrheal illness or skin rash.\nHe had no previously diagnosed long term medical ailments and has not undergone any surgical procedures in the past. He was not on any long term medications and he denied smoking, use of alcohol or illicit drugs. He did not give a family history of any progressive neurological conditions.\nOn general examination he had an average built with no pallor, lymphadenopathy or any signs of malnutrition. No skin rashes or hypopigmented patches were noted. There was minimal small muscle wasting of bilateral hands and feet. No muscle fasciculations were noted. Distal upper limb (hand) power was diminished asymmetrically, right hand demonstrating a power of 3 out of 5 and left hand demonstrating a power of 4 out of 5. All fine finger movements including flexion, extension, abduction and adduction were affected with some degree of weakness in wrist extension as well. Bilateral supinator and biceps reflexes were diminished.\nDistal lower limb (feet) power was also diminished but was less pronounced (power grade 4) when compared to the degree of hand weakness. Bilateral foot dorsiflexion was weak. Ankle jerks were elicited with reinforcement whereas the knee jerks were elicited without reinforcement. There was no objective sensory impairment of touch, pain, temperature, vibration and joint position sensations in both upper and lower limbs. Bilateral plantar responses were down going. No palpable nerve thickening identified. No cerebellar signs were demonstrated and his gait showed a minor degree of high stepping due to weak dorsiflexion. Examination of higher functions and cranial nerves including the fundal examination revealed no abnormality.\nExamination of the cardiovascular, respiratory systems and the abdomen was essentially normal.\nFull blood count revealed white blood cell count: 8.5 × 109/L, platelet count: 274 × 109/L, hemoglobin 12 g/dl with normal red cell indices. Blood picture showed normochromic normocytic cells with some reactive lymphocytes suggestive of a recent viral infection. Serum creatinine 80 μmol/l (60 - 110 μmol/l), serum sodium 138 mmol/l (135 - 145 mmol/l), serum potassium 3.8 mmol/l (3.5 - 5 mmol/l), serum magnesium 0.9 mmol/l (0.8–1.1 mmol/l), serum ionized calcium 1.2 mmol/l (1.05–1.30 mmol/l). Liver profile: AST 21u/l (10 - 40u/l), ALT 13u/l (7–56 u/l), ALP 67u/l (100–360 u/l), serum total bilirubin 0.7 mg/dl (0.1–1.2 mg/dl), serum albumin 36 g/l (35 - 50 g/l), serum globulin 32 g/l (20 - 35 g/l). CPK levels were normal. Inflammatory markers: ESR 25 mm/hour and CRP < 6 mg/dl.\nNerve conduction study revealed findings in keeping with multifocal motor neuropathy with conduction blocks involving the distal upper and lower limb peripheral nerves without any conduction abnormalities in the sensory nerves (Fig. ).\nCSF analysis did not show any increase in proteins or cells and the values were within the normal limits. Anti-GM1 IgM antibody test was not carried out due to the high cost of the test and the patient’s unstable financial background. A sural nerve biopsy (a sensory nerve) was carried out and revealed histologically unremarkable nerve fibres and blood vessels with no evidence of inflammation, atrophy or granulomata formation. Recent dengue infection was confirmed with positive dengue IgM and IgG antibodies with enzyme-linked immunosorbent assay (ELISA).\nAs the patient fulfilled criteria, the diagnosis of multifocal motor neuropathy with conduction blocks was confirmed. He was then referred to the neurologist and was started on intravenous immunoglobulin (IVIg) therapy (2 g/kg/day) which was given for 5 days. He showed a mild improvement of his neurological weakness with the treatment and outpatient physiotherapy was arranged. The next immunoglobulin dose was planned to be given after 2 weeks. |
A seven-year-old girl was referred by her paediatrician with complaint of recurrent pyrexia of untraceable origin. Since low grade chronic dental infections can cause repeated episodes of fever, the paediatrician desired for a dental evaluation. During history taking the patient's mother revealed recurrent episodes of fever ranging from 101 to 102 degrees Fahrenheit with two episodes in a month for the past one year. The fever subsided on taking medication. Adding further she also gave a history of occasional abdominal pain. Upon systemic review with her paediatrician we learnt that she had no history of shortness of breath, chest pain, diarrhoea, rashes, sore throat, ear ache, or urinary symptoms that could indicate the system responsible for such recurrent episodes of pyrexia. The paediatrician also revealed that her chest X-ray and blood profile were normal. There were no abnormalities detected in her urine and stool analysis as well.\nOn examination her facial profile was convex () with normal overjet and overbite. Intraoral examination did not reveal any deep carious lesions or any soft tissue abnormalities or palpable lymph nodes that could have been responsible for recurrent episodes of fever. On taking history for oral habits, the mother stated that the child sucks the great toe of her left feet since infancy (). On examination, the great toe of her left foot was deformed compared to that of the right foot () which led to the diagnosis of toe sucking habit and its possible implication on recurrent episodes of fever. Since the mother and child were supportive to drop the habit, the detailed treatment plan was formulated.\nOn the next visit the parent and the patient were counselled for stopping the habit and they were educated on how it could be deleterious for the developing orofacial structures and on general health of the child. As a reminder therapy, the child was instructed to cover her foot with socks. In the subsequent visit upon finding the patient was reluctant to wear the socks, we implemented the method of negative reinforcement and reminder therapy which was accomplished with a toe guard fabricated with acrylic which fitted the great toe involved in sucking habit ().\nBy three weeks the patient reported reduced frequency of toe sucking habit and episodes of fever. By now the upper incisor showed abnormal pathway of eruption () probably due to abnormal pressure exerted by the toe during sucking event that led to deflection of the developing tooth to an abnormal path. This developing cross-bite was intervened by tongue blade therapy. In the subsequent follow-up visit we noticed that the upper incisors showed distoangular rotation as they erupted into occlusion (). The patient is currently using a preorthodontic trainer (T4K® Phase I, Preorthodontic Trainer for Kids, MRC, Australia) (). The preorthodontic trainer was advocated to realign the incisors as well for myofunctional retraining of the oral musculature.\nAs of now the mother and the child did not report any secondary or substitutional habit after the cessation of toe sucking habit. Also no episodes of fever have been reported. |
A 31-year-old male patient (height 173 cm, weight 70 kg) with no medical history of particular significance had been receiving treatment at a dermatology clinic for ten years for erythematous patches accompanied by non-painful itching in both nasal alar creases, both perioral areas, and both posterior auricular folds (). A topical steroid treatment was chosen, as he had tested negative on the KOH test. The steroid treatment alleviated the symptoms but could not eliminate them completely. It was notable that even on the steroid treatment, skin lesions appeared when he consumed alcohol or was dehydrated. The patient was on the topical steroid treatment when he visited our hospital, and erythematous patches were visible in the nasal alar creases and perioral areas. As a previous study had reported a case of acne vulgaris treatment with SGB [], and the mechanisms of acne vulgaris and seborrheic dermatitis are presumed to be similar, we determined to use SGBs on this patient. An anterior blind approach was selected. The patient was positioned with the anterior spine extended, so that the transverse process of the C6 vertebra (Chassaignac's tubercle) could be easily detected. A needle was vertically inserted at the anterior tubercle and was placed on the transverse process. Five ml of 1% mepivacaine were injected after fixing the needle in position and confirming that there were no abnormal sensations in the upper limb or blood aspiration []. The same procedure was performed daily, on one side each day. After administration of the injection on one side, ipsilateral ptosis and nasal obstruction occurred but faded about two hours later. The skin lesions disappeared within three days, after which the patient wished to stop the treatment for personal reasons. However, the patient revisited the hospital two months later due to a relapse. During the follow-up, the patient noted that there had been no symptoms up to one week before the revisit. We initiated the same treatment procedure and the patient's skin lesions disappeared after five trials (). Fifty more trials were performed, and the patient has not seen a relapse of the symptoms or side effects since. |
A 35-year-old man was hospitalized for acute chest pain and palpitation at rest with a presumed diagnosis of unstable angina. He also described an episode of syncope. He was prescribed chlorpheniramine 4 mg a couple of days ago for symptomatic treatment of upper respiratory tract infection, and he was also on propranolol 20 mg treatment for essential tremor and anxiety disorder for many years. He was used to take propranolol daily. His complaints appeared 1 h after intake of the second dosage of chlorpheniramine together with propranolol. He had no prior history of cardiac arrhythmias, valvular or congenital heart disease, and sudden cardiac death in family history. He smoked regularly up to 20 cigarettes a day. He denied the usage of legal or illegal other medications such as antidepressants and cocaine. Physical examination revealed irregular pulse, low blood pressure of 90/60 mmHg, and altered cloudy consciousness. He had no biochemical abnormality; magnesium and potassium level was in normal range. Electrocardiography (ECG) showed ST-segment depression in inferior leads and isolated ST elevation in leads D1-aVL []. Echocardiography showed normal systolic functions with no segmental wall motion abnormalities. The patient was given 300 mg of acetylsalicylic acid and underwent emergency coronary angiography that revealed normal coronary arteries. Whenever ischemic changes were observed in admission ECG, anti-ischemic therapy was not admitted to the patient due to the result of completely normal coronaries. After all the patient was followed by telemetry at coronary care unit. Control ECG showed ventricular bigeminy and R on T phenomenon. On the 2nd day of admission, the patient had polymorphic ventricular tachycardia (VT) attack which degenerated to ventricular fibrillation, immediately patient was defibrillated and sinus rhythm was restored successfully []. After defibrillation, the patient continued to have frequent premature ventricular beats and couplets on the monitor. On the 3rd day of his admission, an upper rib ECG was performed and showed a pattern of right bundle branch block with ST-segment elevation in leads V3. The present findings were thought to be compatible with Type 3 Brugada syndrome []. An ajmaline challenge test[] was then performed. Although the frequency of ventricular extrasystoles and couplets was increased during the test, no coved-type ECG pattern appeared in the right precordial leads []. Therefore, the diagnosis of Brugada syndrome was abandoned. Electrophysiological study was proposed, but the patient refused the test. Because the patient survived a sudden unexplained cardiac arrest, it was decided to implant an implantable cardioverter defibrillator for secondary prevention. The patient recovered and had no further complaints after implantation and he was discharged. |
A 69-year-old male was admitted to a hospital in June 2016 because of right arm asthenia and dysarthria and was diagnosed as having cerebral infarction in the left middle cerebral artery area along with deep vein thrombosis. ECG demonstrated normal sinus rhythm and echocardiography revealed no intra-cardiac thrombus or vegetation. The patient was discharged from the hospital following administration of apixaban.\nIn August 2016, the patient was readmitted to the hospital because of recurrent right arm asthenia and dysarthria. MRI revealed multiple cerebral infarctions in not only the bilateral cerebral hemispheres but also the cerebellum. Trousseau’s syndrome was suspected at this time. Apixaban administration was stopped and an intravenous drip of heparin was started. Echocardiography revealed mild mitral regurgitation with vegetation on the mitral valve. Although the laboratory data suggested no evidence of infection, ceftriaxone and gentamicin were added as a precaution against infective endocarditis. The patient was then referred to our hospital for surgery.\nA CT scan revealed a left renal infarction and multiple swollen lymph nodes around both the abdominal aorta and stomach with antral hypertrophy, suggesting an advanced gastric cancer or lymphoma. As the vegetation showed no change despite the heparin and antibiotics therapy, cardiac surgery was performed on day 5 after referral. Extracorporeal circulation was instituted employing aortic and bicaval cannulation. After aortic cross-clamping, the mitral valve was exposed via a left atriotomy. Both mitral leaflets had vegetation on the surface, and major vegetation 15 mm in width was evident on the anterior leaflet (Fig. ). These were resected in their entirety and replaced with a 25-mm Epic bioprosthesis (Abbott). Continuous intravenous heparin administration was resumed on the following day, aiming for an activated partial thromboplastin time of between 40 and 50 s. Histologic analysis revealed that the vegetations were thrombi covered with vascular endothelium and that the mitral leaflet tissue was not damaged (Fig. ). On the basis of these findings, the patient was diagnosed as having NBTE.\nAn endoscopic stomach biopsy was performed on the seventh postoperative day, and histologic analysis revealed non-solid poorly differentiated adenocarcinoma with components of signet-ring cell carcinoma and moderately differentiated tubular adenocarcinoma. The patient was definitively diagnosed as having Trousseau’s syndrome and, subsequently, transferred to the department of surgery. A Billroth I distal gastrectomy was performed, and a continuous intravenous heparin drip was employed during the operation. Histologic analysis revealed poorly differentiated adenocarcinoma with a component of moderately differentiated tubular adenocarcinoma and metastatic tumor cells in the dissected lymph nodes (T4aN3bM0; stage IIIb). Further histologic analysis using alcian blue staining confirmed the presence of mucin in the tumor.\nSubcutaneous heparin injection was introduced on day 8 after the gastric surgery, and the patient was discharged from our hospital in October after acquisition of the self-injection technique. During this long hospitalization, no thromboembolic events were observed. Chemotherapy was started in November. The patient has survived for 18 months after the diagnosis of Trousseau’s syndrome without any recurrence of thromboembolism. |
A 40-year-old Indian female consulted our oral and maxillofacial clinic, complaining of discomfort during swallowing. The patient was apparently alright two months back when she developed mild discomfort during deglutition. Around the same time the patient also noticed a painless swelling over the upper left side of the neck, which gradually kept increasing in size. No significant medical history or history of previous trauma was reported. On extraoral examination, no gross facial asymmetry was noticed. Intraoral examination showed upper and lower jaws with normal teeth. No other mucosal abnormality was detected over oral and oropharyngeal mucosa. Palpation of the inferior border of the mandible revealed a painless bony hard mass over the inferior border of the mandible in relation to the left canine‒premolar region. The growth was fixed to the inferior border and lingual surface of the mandible and did not show any movement during deglutition (). Cervical lymph nodes were not palpable.\nAn orthopantomogram showed a well-defined homogenous radiopaque mass arising from basal part of the mandible below the apices of the left premolar and the first molar teeth with a continuous contour (). Computed tomography (CT) scan with sectional and 3D formatted images was obtained for further evaluation of the lesion. CT scan showed a well-defined, hyperdense mass measuring 2.5×2 cm in size, arising from the left lower border of the mandible. Coronal and sagittal sections showed the growth projecting slightly towards the lingual aspect and impinging over the muscle plane on the oral cavity floor and neck (). No other lesion was seen involving the craniofacial skeleton. Based on the clinical and radiological findings, provisional diagnosis of peripheral osteoma was made.\nSurgical excision of the tumor was planned and executed under general anesthesia. The surgical approach used was submandibular incision placed in the upper neck crease. Upon layered dissection over the lower border of the mandible, the anterior belly of the digastric muscle was seen overriding the lesion on its buccal aspect (). The muscle was retracted medially by blunt dissection. The lesion was isolated from surrounding muscles and excised. The excised specimen was submitted for histopathological analysis. Microscopic sections of the specimen showed compact bone having mature lamellae with scanty marrow (). Based on clinical presentation, imaging characteristics and histological features a final diagnosis of solitary peripheral ivory osteoma of the mandible was reached. The patient was recalled at regular intervals and showed complete resolution of symptom. A follow-up panoramic view taken at one-year postoperative interval showed complete healing of the cortical aspect of the inferior border of the mandible with no sign of recurrence (). |
A 58-year-old Korean woman was referred to our institution for a mass in the right distal tibia detected with magnetic resonance imaging performed at an outside clinic (). The patient had been suffering from pain in her right ankle for a week. An open biopsy was performed, and the tumor was pathologically diagnosed as an osteoblastic conventional osteosarcoma (). She was transferred to our institution, where she subsequently underwent chest radiography and computed tomography (CT) for routine baseline evaluation. Although there was no evidence of pulmonary metastasis, two calcified nodules were discovered, one in the right breast and the other in the anterior mediastinum (). Based on imaging features, benign granuloma and metastasis were considered in the differential diagnosis, and short-term follow-up was recommended because the patient was scheduled for neoadjuvant intra-arterial chemotherapy. Chest CT performed three weeks later revealed size increases of the calcified masses (), raising the suspicion for metastasis. Therefore, the patient was referred to the breast radiologist for biopsy.\nDiagnostic mammography () showed a dense calcified mass in the right upper breast, with spiculated margins. Breast ultrasonography (US) of the right upper breast () revealed a 1.5 cm dense calcified mass with posterior shadowing. Owing to the dense calcifications seen in the mass, percutaneous US-guided core needle biopsy was considered difficult to perform, and excisional biopsy was recommended to make a pathologic diagnosis. After the third cycle of neoadjuvant chemotherapy, excisional biopsies of the breast and mediastinal masses were performed. Specimen mammography () of the excised breast mass showed the dense calcified mass within the specimen, confirming complete excision of the mass. Pathologic examination revealed a high-grade spindle cell tumor with osteoid matrix production (). Immunohistochemical staining revealed positivity for cluster of differentiation 99 (CD99) and negativity for estrogen receptor (ER), progesterone receptor (PR), and basal cytokeratins (CK5/6), which further suggested metastatic osteosarcoma to the breast (). The mediastinal mass was also excised via video-assisted thoracoscopic surgery, confirming metasta-tic osteosarcoma. Currently, the patient is being treated with adjuvant chemotherapy, with no clinical or radiological findings of recurrence after 2 months of follow-up. |
A 34-year-old male patient was referred to the Oral and Maxillofacial Surgery Department for the treatment of a calcifying odontogenic cyst that was localized in the right maxilla. He complained of a slow, progressive and painless growing lesion during the last 3 years. Physical examination revealed a bulging and hard mass, slightly effacing the right nasogenian fold. Intraoral examinations disclosed the expansion of the vestibular and palatal cortical bones ().\nThe computerized tomography (CT) showed a large expansive lesion, with soft tissue attenuation, occupying the totality of the right maxillary sinus, reabsorbing the sinusal walls and obliterating the maxillary sinus ostium ().\nDue to the lesion size and its proximity to the lateral wall of the nasal cavity, orbital floor, and pterygopalatine fossa, it was was first decompressed under local anesthesia, what also rendered specimens for histological analysis. The cystic cavity was opened towards the oral cavity, and was left open until the second surgery, which was accomplished 9 months later ().\nClinically, 3 months after the decompression, one was noticed partial regression of the lesion ().\nCT scan 5 months after the decompression, also demonstrated satisfactory regression of the lesion (). A new surgical approach under general anesthesia was undertaken, allowing total excision. In this procedure, the maxillary sinus ostium could be maintained, keeping adequate sinus drainage.\nThe anterior wall of the right maxillary sinus and the former decompression orifice were exposed followed by an ostectomy to enlarge the access to the cyst contents.\nThe whole lesion was more easily excised due to a defined cleavage plan between the tumor and the remaining bone. Additional curettage was performed. Dental elements 12 and 13 were removed as no surrounding bone was present. With the aim of maintaining normal sinus drainage, a perforation was made to communicate the maxillary sinus to the nasal cavity through which a fine silicone probe was inserted and maintained by being stitched to the right nostril for 20 days ().\nThe gross view of the specimen post-surgical excision is demonstrated in .\nHistological examination of the surgical specimen showed a fibrous cystic capsule with epithelial lining showing columnar basal cells, resembling ameloblasts, with somewhat loose upper layers containing isolated and clustered ghost cells, consistent with the diagnosis of CCOT ().\nThe postoperative period was uneventful. Thirteen months after the full excision, the patient was completely recovered with no clinical signs of recurrence. At intraoral examination, there was still a cortical expansion of vestibular and palatal aspects of the right maxilla, but in a smaller size ().\nCT scans obtained 13 months after surgery showed bone formation inside the maxillary sinus as well as a radiolucent margin, suggesting the proliferative potential of healthy remaining sinus lining. Long-term annual clinical and imaging controls are expected in order to detect eventual recurrences (). |
A 57-year-old asymptomatic man with no significant past medical history was found to have an enlarged cardiac silhouette on a routine chest radiograph (). Magnetic resonance imaging (MRI) revealed a 9 cm pericardial cyst in the right cardiophrenic angle that was associated with right atrial compression (Figures , , and ). Although the pericardial cyst wall showed contrast uptake, no uptake within the cyst was observed on first-pass or delayed images. There was no compression of the airway or superior vena cava (SVC) and the pericardial cyst had not eroded into the heart. The patient was not at high risk for hydatid cysts and he did not have any history of fever, suggesting that an infectious cause for his pericardial cyst is unlikely. He did not have any history of chest trauma or intrathoracic surgery. The absence of hypertension, hematuria, and a positive family history made a diagnosis of autosomal dominant polycystic kidney disease (ADPKD) unlikely. The patient was scheduled for resection of the pericardial cyst using VATS. Preoperative electrocardiographic findings, complete blood count results, serum creatinine levels, liver function tests, and serum electrolyte levels were normal.\nOn the day of surgery, the physical exam, including heart and lung auscultation, was unremarkable and the vital signs were within normal limits (blood pressure of 119/75 mmHg, heart rate of 83 beats per minute, respiratory rate of 14 per minute, blood oxygen saturation of 97% on room air, and temperature of 36.9°C). A left radial arterial line and two large-bore intravenous catheters were placed. The patient was adequately hydrated with intravenous administration of normal saline. He was transferred to the operating room and placed in the supine position on the operating table. The standard American Society of Anesthesiologists monitors were placed on the patient. The pericardial cyst did not compress the patient's right bronchus or the SVC, and therefore, he was able to tolerate the supine position with no shortness of breath or hemodynamic instability. The patient was preoxygenated and general anesthesia was induced by slow intravenous administration of etomidate 0.2 mg/kg and fentanyl 1 μg/kg. Neuromuscular blockade was achieved by intravenous administration of succinylcholine 1.5 mg/kg. Ephedrine 0.1 mg/kg was administered following induction to minimize the hemodynamic effects of the induction agents and positive pressure ventilation. A 37-French left-sided double-lumen endobronchial tube (DLT) was placed. The patient was placed in the left lateral decubitus position. Anesthesia was maintained with oxygen (fraction of inspired oxygen of 0.6), air, and sevoflurane (1 minimum alveolar concentration). The right lung was collapsed and neuromuscular blockade was induced with rocuronium 0.6 mg/kg. A 1-cm incision was made in the posterior axillary line at the fifth intercostal space. A metal port was placed and a 10-mm, 30° thoracoscope was placed through the incision. A 2-cm incision was made above the fifth intercostal space at the mid axillary line to access the cyst. The pericardial cyst was found to be firm, and had some calcifications on the surface (Figures , , and ). The cyst was easily separated from the pericardial fat. However, it was attached to the anterior chest wall. Following separation of the cyst from the anterior chest wall some bleeding occurred from the distal right internal mammary artery. The bleeding was controlled with Enseal and clips. The cyst was large, and hence, access to the superior aspect of the cyst was difficult. Therefore, an 18-gauge needle was used to aspirate the cyst fluid. Approximately 300 ml of brown, murky, nonodorous fluid was aspirated from the cyst before it was completely resected (). The patient remained hemodynamically stable throughout the procedure and the DLT was removed in the operating room at the end of the procedure. Postoperative pain was managed with an intercostal nerve block using 10 ml of 0.5% bupivacaine and patient controlled analgesia pump using hydromorphone (intravenously, 0.2 mg every 10 minutes). The patient's postoperative course was uneventful, and he was discharged on postoperative day 1 in a stable condition. Cyst fluid cultures were negative. |
A 61-year-old woman was referred to our center for urgent coronary angiography due to failed thrombolysis and ongoing chest pain. She had been previously admitted to a hospital in another city far away from our center due to acute inferoposterior myocardial infarction (MI). She had no known atherosclerotic risk factors, but laboratory examination revealed dyslipidemia with high serum triglyceride (230 mg/dl) and low high-density lipoprotein cholesterol (36 mg/dl). On admission, hemodynamics was stable and chest pain had subsided. On electrocardiography, there were low-voltage limb leads with small q waves in the inferior leads and a pattern of incomplete right bundle branch blockage with an R/S ratio greater than 1 in lead V3 in the precordial leads, altogether indicating a recent inferoposterior MI. Echocardiography revealed inferoposterior wall akinesis with a left ventricular ejection fraction of 40%.\nThe patient subsequently underwent coronary angiography. The left circumflex artery (LCx) was cut off after the first obtuse marginal. The LAD had severe long stenosis (up to 90%) at midportion. An anomalous vessel was seen originating from the midportion of the LAD (shortly after the second septal perforator within the diseased segment), which followed the course of the RCA (). In this case, the anomalous RCA traveled along the free wall of the right ventricle into the atrioventricular groove and gave rise to a posterior descending artery (). Interestingly, there was also a separate small artery originating from the right coronary sinus that was most probably a right atrial branch (). Aortic root injection confirmed the absence of the RCA stump or any other vessel except for the small right atrial branch in the coronary sinuses as well as the ascending aorta ().\nA decision was made to predilate the LCx lesion using a 1.5 × 15 mm balloon in order to visualize the distal bed. There were 2 more large obtuse marginal branches at distal (). Stenting was not performed due to poor antegrade flow and heavy atherothrombotic burden. The patient was put on intravenous Eptifibatide and Heparin and discharged four days later on Aspirin, Clopidogrel, Metoprolol, Captopril, Atorvastatin, and nitrates. Unfortunately, she did not return for follow-up for further percutaneous or surgical revascularization. |
A 65-year old female of histopathologically proven primary nasopharyngeal carcinoma was treated with concurrent chemo radiation of 60 Gy radiotherapy in 30 fractions of 2 Gy each, along with weekly Cisplatin. Except for well controlled type 2 diabetes on oral hypoglycemic drugs, the patient had no other co morbid conditions, and was symptom free after completion of the radical treatment. The clinical examination revealed no evidence of disease at the local site and no loco regional lymphadenopathy. Hematological, biochemical and metabolic parameters were within normal limits. F18 FDG PET/ CT was performed for the end of treatment evaluation and compared with the pretreatment 18F-FDG PET/CT. Present study revealed no abnormal morphologic or metabolic focus at the primary site of nasopharynx and no loco regional adenopathy either morphologic or metabolic. Interestingly, in the abdominal sections, a large heterogeneous peripherally enhancing hypodense 85x83x80 mm mass with peripheral FDG avidity and central void lesion containing multiple pockets of air lucencies within, was seen in the postero inferior aspect of right kidney [] within feromedial infiltration into the adjoining right psoas muscle []. The free gas lucency was also noted in non dependent part of the urinary bladder and hypodense debris amidst the contrast filled bladder at the right vescico ureteric junction []. The findings were characteristic of emphysematous pyelonephritis. Ultrasonography (USG) revealed classical echogenic scattered echoes typical of free gas in a mixed echoic mass corresponding to the PET CT outlined lesion in the right kidney []. The initial PET CT done prior to initiation of treatment was reviewed to see the renal morphology, which showed both kidneys of normal morphology and excretion []. In view of the potentially serious nature of the condition and the patient being absolutely symptom free and afebrile, a detailed retrospective history was taken, which revealed an episode of febrile neutropenia during the fourth cycle of chemotherapy. It was managed conservatively and the patient was afebrile since then. Despite the present symptom free state, since the condition can flare up any time, a curative treatment was planned whenever immune status was compromised. The conventional options were nephrectomy and percutaneous drainage, the former radical surgery and the latter invasive. In view of the evidence of free gas in the bladder and the debris seen at the vescicoureteric junction, there was an ample objective proof of the abscess communicating with collecting system. Hence, a Double J (DJ) stent placement to facilitate better drainage of infective material into the bladder alongwith broad spectrum antibiotics was endeavored before embarking on more invasive and radical measures. The patient was followed up after three weeks with an ultrasonogarphy, which showed gratifying near complete disappearance of the abscess, and return of the renal contour to normal and disappearance of all the gas in the kidney and bladder as well []. Plain CT and topogram of abdomen subsequently revealed disappearance of free gas lucencies in the right renal region with DJ stent in situ [] with resolution of the emphysematous abscess, urinary bladder gas and debris []. The patient on follow up was disease free from both the primary nasopharyngeal carcinoma and emphysematous pyelonephritis. |
A 37-years-old male presented to UiTM Primary Care Clinic, Sungai Buloh, Selangor, Malaysia with a history of persistent left hip pain. The patient had underlying bronchial asthma, present since childhood, that had been well-controlled with salbutamol use as required. The patient was not on any inhaled corticosteroids.\nThe patient first presented with a 2-week history of left anterior hip pain that occurred after he slipped on the stairs and landed on his left knee with no direct trauma to the hip. The pain worsened with initiation of movement and weightbearing on the affected joint, especially when exiting a car or climbing stairs. He was overweight, normotensive, with full range of movement of the bilateral hips. The patient had no history of prolonged corticosteroid use, alcoholism, and he had no systemic medical conditions. Left hip X-ray showed no obvious evidence of fracture and he was discharged with analgesia. It was difficult to attribute the fall as a cause for the hip pain since there was no direct trauma to the hip; however, the hip pain could be a referred pain from the knee.\nThe patient then presented a month later complaining of acute lower back pain which radiated to the left groin and had been present for 3 days. He was noted to have an antalgic gait with paravertebral muscle spasm. On examination, the straight leg raise test was negative with normal neurological examination of the lower limbs. Lumbosacral X-ray was normal and the patient was discharged with analgesia. He was reviewed 2 days later, stating improvement and no longer had an antalgic gait.\nSubsequently, the patient continued to have several recurrent episodes of intermittent left hip pain. In between attacks, his pain would partially subside. He returned to the clinic with similar symptoms and this prompted the primary care team to arrange for an orthopedic assessment. On examination, the patient had limited internal rotation and external rotation of his left hip and a repeated left hip X-ray showed sclerotic changes (). In view of these findings, a magnetic resonance image (MRI) of bilateral hip joints was arranged (). The results revealed bilateral femoral head avascular necrosis with left hip synovitis. This was classified as Association Research Circulation Osseous stage II.\nThe patient underwent core decompression of the left hip and an autologous iliac bone graft around 5 months after his first point of care assessment. His right hip was treated conservatively as it was asymptomatic and the changes from the MRI were minimal. Histopathological examination of the left femoral head biopsy showed necrotic bony tissue with no malignancy features seen (). The patient was discharged on oral paracetamol and tramadol and was put on strict non-weight bearing crutches.\nThe patient attended regular follow-up appointments following the surgery and his progress was monitored according to pain control and hip X-ray examinations. In general, his pain score was mild, and his serial X-rays showed good graft incorporation; however, sclerotic changes were seen. He was subjected to hip abductor strengthening exercises with gradual ambulation until he was able to achieve full weight bearing.\nAll authors declare that written informed consent was obtained from the patient for publication of this case report and accompanying images. |
The patient was a 36-year-old gravida 0 woman. At the age of 7, she underwent ventricular septal defect closure for the right ventricular outflow tract. At the age of 11, she received a mechanical aortic valve replacement. Since after the replacement, she has been receiving warfarin orally at a dosage of 4.5 mg/day. She conceived naturally and she was referred to our hospital for perinatal management. Oral administration of warfarin was discontinued at 5 weeks of gestation and she began self-injection of heparin. At 21 weeks and 5 days of gestation, she was admitted to our hospital with a high risk of spontaneous abortion and was put on intravenous ritodrine. This successfully prevented a miscarriage. At 21 weeks and 6 days of gestation, we started a continuous infusion of 25,000 units of heparin daily. On the 22nd week, transesophageal echocardiography showed a movable thrombus in the aortic valve. The size of the biggest thrombus was 26 × 8 mm (). We increased the dosage of heparin to 28,000 units daily and restarted the administration of warfarin. Following this, the thrombus reduced in size, and at 23 weeks and 5 days transesophageal echocardiography showed no signs of thrombosis in the patient. At 32 weeks and 2 days of gestation, a routine cardiotocography showed a decreased fetal heart rate; thus, an emergency Cesarean section was performed under general anesthesia because of the presence of warfarin in the blood. The baby was delivered, weighing 1,702 g, with an Apgar Score of l at l minute, and 4 at 5 minutes. The total amount of blood loss during the surgery was 1,410 ml. During the surgery, 16 units of fresh frozen plasma (FFP) was transfused; and after surgery, we continued to infuse 20,000 units of heparin daily. On the 11th day after surgery, owing to continuous genital bleeding, heparin administration was discontinued and uterine artery embolization was performed. This treatment stopped the bleeding and on the 21st postsurgical day; we started warfarin administration at 5 mg/day. She was discharged on the 34th postoperative day due to the stable PT-INR levels ().\nThe newly born infant was intubated and admitted to the newborn intensive care unit. At the time of admission, activated partial thromboplastin time was 180 seconds or more and bilateral intracerebral ventricular hemorrhage was detected using ultrasonography. On the first day of life, anemia was observed in the infant and red cell concentrate and FFP were transfused (). We attempted to reduce the infant's dependence on the ventilator and at 8 days of age the infant was extubated. On the postnatal 10th day, a cranial CT scan showed bilateral intraventricular hemorrhage with ventricular dilation and midline shift (). Although convulsions accompanying the intracranial hemorrhage were observed, the infant's general condition was stable and oral feeding was started on postnatal day 10. The newborn was discharged on postnatal day 54. However, the infant later developed cerebral palsy and is currently receiving treatment at our hospital. |
A 59-year-old woman was admitted to the emergency room of a local hospital with the sudden onset of severe chest pain radiating to her back. She had a history of hypertension and a cerebrovascular accident. At the time of presentation, she had a drowsy mental status. The cardiac enzymes were elevated on baseline laboratory testing. A chest film showed mediastinal widening (). The initial electrocardiogram (ECG) showed a normal sinus rhythm without significant ST-T changes (). An emergent chest CT scan was performed under the suspicion of an acute aortic dissection, which showed an acute Stanford type A aortic dissection (). The patient was then transferred to our institution for repair of the aortic dissection. She was hemodynamically unstable.\nEmergent ascending aortic replacement surgery using a graft was performed with the patient under general anesthesia using a median sternotomy. Upon opening the pericardium, a hemopericardium was noted. The aortic root was incised, and an intimal tear was identified just above the sinus of Valsalva. The dissection had not extended to the orifice of the left coronary artery. We carried out an ascending aortic replacement with a 28-mm Hemashield® graft. Systemic circulation was then restarted. ST segment elevation was observed on the ECG monitor immediately postoperatively. Sedation and mechanical ventilation were maintained, and she was transferred from the operating room to the intensive care unit (ICU).\nIn the ICU her blood pressure and cardiac output remained low in spite of sufficient inotropic support. An ECG showed ST segment elevation in leads II, III, and aVF, and in the precordial leads, suggesting broad myocardial ischemia (). In addition, transthoracic echocardiography showed regional wall motion abnormalities in the territories of the left anterior descending artery (LAD) and left circumflex artery (LCx), and moderately decreased LV systolic function. These results suggest coronary artery malperfusion, probably caused by progression of the aortic dissection into the left coronary artery. Accordingly, emergent coronary angiography was performed 90 minutes postoperatively.\nCoronary angiography revealed no significant luminal narrowing in the right coronary artery, but almost complete collapse of the lumen of the LAD (). After forceful contrast injection into the LAD, the collapsed lumen was reopened, but it subsequently recollapsed without forceful contrast injection. A similar finding was observed in the LCx (). We reasoned that this life-threatening coronary artery occlusion was caused by pulsatile compression of the false lumen of the left coronary artery dissection. A zotarolimus-eluting stent was deployed in the mid-portion of the LAD and a bare metal stent (Tsunami®, 3.5×30 mm) was deployed in the proximal portion of the LAD. Three bare metal stents (Driver®, 3.0×30 mm; Tsunami®, 3.0×30 mm; and Driver®, 3.0×18 mm) were successfully deployed from the distal portion to the proximal portion of the LCx. However, coronary blood flow was not improved. Thus, direct stenting at the left main stem, and a bare metal stent (Driver®, 3.5×18 mm) was successfully deployed. The final angiogram of the left coronary artery showed Thrombolysis In Myocardial Infarction (TIMI) flow grade 3 in the LAD and LCx.\nFollow-up coronary angiography was performed 45 days after percutaneous coronary intervention (PCI) because the patient remained unstable. Dyspnea was New York Heart Association (NYHA) class III/IV and follow-up two-dimensional echocardiogram (2D-UCG) revealed a severe decrease in LV systolic function. Follow-up coronary angiogram showed significant in-stent restenosis at the proximal portion of LAD (). An intravascular ultrasound (IVUS) study showed the remaining coronary artery dissection in the diagonal branch of the LAD (). Repeat balloon angioplasty using a 4.0×20 mm balloon (Apollo®) was performed at the site of in-stent restenosis. The final angiogram of the LCA showed TIMI flow grade 3 in the LAD and LCx. Her clinical course following the repeat PCI was stable and uneventful. The patient was discharged from hospital 51 days after the surgical repair. A follow-up coronary angiogram was performed 8 months after the PCI and showed no significant in-stent restenosis (). |
A 40-year old infertile woman was referred to our center for IVF program (oocyte donation and embryo transfer). She had two previous histories of dilatation and curettage, one after spontaneous abortion and another for diagnostic purposes.\nHysteroscopy was done prior to IVF cycle which revealed a band of adhesion running from anterior to posterior wall for which adhesiolysis was done in the same session. The patient conceived in the second cycle of embryo transfer (oocyte donation). Pregnancy was confirmed by beta-human chorionic gonandotrophin (β–hCG) levels and later on, by ultrasound which confirmed single intrauterine gestational sac with a beating heart. She was followed up regularly for her obstetric examination with no other risk factors except for advanced maternal age.\nLevel two obstetric ultrasound examination was done at eighteen weeks of gestation which ruled out gross congenital anomalies and suggested grade 0 left fundo-anterior placenta with normal invasion and normal amniotic fluid. In the nineteenth weeks of gestation, patient presented in the emergency department of our hospital with acute abdominal pain and signs of early shock (pulse rate 180/min, blood pressure 90/60 mmHg, hemoglobin 7.5 g% and hematocrit of 24%).\nInitially, the patient was revived from shock with adequate fluid replacement and other basic management procedures including central line placement for cental venous pressure (CVP) monitoring. Once the patient's condition was stable, she was sent for ultrasound and CT scan evaluations. Ultrasound examination revealed fresh intrauterine fetal death along with hemoperitoneum with no other gross finding.\nCT scan was done to rule out other causes of heamoperitoneum. Findings were suggestive of weakening of the uterine musculature at the left fundal region with fetus inside the cavity. Patient was taken up for emergency laprotomy and a heamoperitoneum of 3000 ml was found along with a dead fetus coming out of a large uterine fundal defect. Female fetus of approximately 700 g was extracted but the placental tissue could not be separated from the myometrium. Subtotal hysterectomy was performed in view of massive haemorrhage, while all other intraperitoneal organs were found to be intact. The patient was transfused one unit of packed cell intraoperatively and two units thereafter. She was intensively monitored postoperatively and was discharged on the sixth post-operative day in a perfectly stable condition.\nUterus () sent for biopsy and pathology report showed chorionic villi reaching the myometrium with absent decidua basilis which were consistent with the diagnosis of placenta accreta (). |
An 84-year-old female with a known history of congestive cardiac failure, hypertension, and stable angina was initially seen in the Emergency Department at Horton General Hospital, Banbury. She presented with an acute onset of rapidly worsening exertional dyspnoea and reduced exercise tolerance. She was subsequently diagnosed with critical aortic valve stenosis following transthoracic echocardiography and underwent an urgent aortic valve replacement. The immediate postoperative course was uneventful except for the development of a pericardial effusion requiring drainage on the 10th day. Microbiological analysis of the pericardial fluid yielded cultures of the gram-negative pathogen, Proteus mirabillis. The patient completed a full course of intravenous gentamicin for 3 weeks with serial negative blood cultures. The wound site appeared to be healing well throughout this time. Further transesophageal echocardiography revealed no significant pericardial effusion and the patient subsequently made a good recovery and was discharged. Further cross-sectional imaging was not performed at this time.\nFour weeks following discharge from hospital (two months postoperatively), the patient represented with fever, worsening dyspnoea at rest, and light headedness. She was clinically in septic shock and physical examination revealed an erythematous, warm anterior chest wall alongside a fluctuant swelling overlying the sternotomy scar with some purulent discharge. Laboratory data included a severe normocytic anaemia, neutrophilic leukocytosis, and significantly raised inflammatory markers. Subsequent blood cultures grew meropenem sensitive Proteus mirabillis.\nInitial treatment against the bacteraemia consisted of intravenous gentamicin and meropenem based on a working diagnosis of mediastinitis with or without bacterial endocarditis. The chest radiograph was unremarkable. Subsequent chest CT revealed osteomyelitis of the sternum, contiguous with a large anterior mediastinal abscess measuring 67 mm by 44 mm in diameter (). The more interesting radiological feature was the direct communication and erosion of the abscess cavity into the anterior ascending aorta resulting in a localised periaortic area of contrast extravasation (). The concomitant presence of endocarditis was ruled out on echocardiography which also revealed the large mediastinal cavity in close proximity to the aortic root as well as a 1.4 cm diameter pseudoaneurysm in the anterior ascending aorta.\nPeridiagnosis, there were marked haemodynamic instability and a rapid decline. In view of her comorbidities, surgery was not deemed a suitable option and consequently the patient died soon after with pseudoaneurysm rupture as the attributed cause of death. |
We present the case of a 45-year-old Caucasian man suffering from psoriasis for more than 20 years at the time of the first visit. No relevant comorbidities were found at history collection, apart from benign prostatic hyperplasia, for which he is treated with tamsulosine. He was initially diagnosed and followed-up by dermatologists, who treated him with topical corticosteroids and salicylic acid for skin involvement and systemic corticosteroids for resistant lesions as needed ().\nThe skin component had always been <30% of the total body surface area (BSA). The patient reported distress in personal and social relationships with consequently reduced HRQoL. After ten years, he started experiencing dactylitis of feet and recurrent arthritis of the small joints of hands, shoulders and knees, together with inflammatory morning stiffness lasting around one hour, with negative rheumatoid factor (RF); thus fulfilling ClASsification criteria for Psoriatic ARthritis CASPAR criteria for PsA []. He was referred to the rheumatology outpatient clinic of another hospital and treated with indomethacine and systemic corticosteroids as needed for about five years, and then achieved complete remission of joint symptoms. Three years later, because of a flare of joint involvement (episodes of peripheral arthritis and dactylitis of the small joints of hands and feet once per week) and skin involvement, he was started on methotrexate 10 mg weekly with folate supplementation, in combination with cyclosporine 200 mg daily, with reduced extension and severity of skin lesions and decreased frequency of arthritis flares to once per month. However, he did not reach complete remission. Shortly after initiation, due to a 2-fold increase of liver enzymes (hepatitis excluded based on abdomen ultrasound and hepatitis B and C viral profile), methotrexate was reduced to 7.5 mg weekly []. The reduced methotrexate regimen led to normalization of the liver enzymes but was ineffective in controlling both skin and joint involvement. Two years later, due to elevated arterial pressure values (up to 150/90 mmHg) and serum creatinine increase >30% of baseline value (up to 1.4 g/L), cyclosporine was stopped.\nThe following year, he referred to our outpatient clinic for skin and joint flare. At referral, physical examination demonstrated mild psoriasis of trunk, arms and legs, with a psoriasis area severity index (PASI) score of 5.1 (), dactylitis of the third digit of the left foot and arthritis of the third interphalangeal joint of the left hand, which, together with a normal C reactive protein (CRP), activity visual analogue scale (VAS) and pain VAS of 4 and 4.5 respectively, accounted for a disease activity in psoriatic arthritis (DAPSA) score of 12 (low disease activity) [].\nLaboratory tests showed no relevant alterations. Radiographic assessment of hands and feet showed no erosions and no signs of axial involvement were found at magnetic resonance imaging (MRI). Ultrasound imaging of the third metacarpophalangeal (MCP) joint showed inflammation of the joint and of the peritendon of the extensor tendon (a).\nConsidering that the patient failed to respond to two DMARDs (cyclosporine and methotrexate), that he refused a parenteral drug, that he presented mild skin and joint involvement, no bone erosions, dactylitis and no axial involvement, apremilast was chosen [,,]. At baseline, the EQ-5D questionnaire reported altered HRQoL, with some difficulties in motility, no difficulties in self-care and some difficulties in usual activities, moderate pain/discomfort and moderately anxious/depressed mood. Patient’s global health (GH) VAS was 60 and pain VAS was 45 (). Furthermore, the patient was screened at baseline for CV risk; carotid arterial doppler ultrasound demonstrated only intimal thickening of the left bulb and 24 h blood pressure monitoring demonstrated mild systolic and diastolic daily hypertension for which he started lercanidipine 10 mg/daily. At baseline, total cholesterol (TC) and triglyceride (TG) serum levels were 173 mg/dL and 113 mg/dL respectively, and TC/high density lipoprotein (HDL) ratio was 4.94 (). After 4 weeks, the extension and severity of erythema and infiltration of psoriatic plaques were substantially reduced (PASI 2.5, ). Furthermore, the patient reported a marked improvement of joint involvement with no further episodes of arthritis or dactylitis, although inflammatory morning stiffness persisted (DAPSA 6.0). At the 2-month follow-up, the patient reported several side effects such as mild headache, dizziness and hypotension and, due to further amelioration of the skin and joint involvement he stopped apremilast, which was restarted shortly after, due to worsening of psoriasis (PASI 3.0) and occurrence of a new joint flare (DAPSA 10). At the time he was still taking methotrexate 7.5 mg weekly. At the 6-month follow-up he reported no arthritis flares and no inflammatory morning stiffness. At physical examination, the third MCP joint was swollen but not tender (DAPSA 3), and ultrasound imaging showed reduction of joint and soft tissue inflammation (b). A further improvement of skin involvement was observed and reported by the patient (PASI 1.2, ). During the follow-up, we observed an amelioration of the serum lipid profile already after 4 weeks, with a 5.8% reduction of TC and a reduction of TC/HDL ratio, and after 12 months, a further reduction of TC, LDL and TG values of 15.6%, 25.7% and 17.7% respectively, and a 20% increase of HDL levels compared to baseline values. TC/HDL ratio reached the favorable value of 3.48, as shown in . We even observed a 5% weight loss—although our patient already had a normal baseline weight and body mass index (BMI)—reaching a final normal weight with a normal body mass index (BMI 21.5). After 12 months, the clinical manifestations were stable. We therefore decided to stop methotrexate, but the patient experienced a slight relapse of both skin and arthritis (PASI 2.5, DAPSA 6), shortly after. Therefore, although dosage was low (7.5 mg), we reintroduced methotrexate, leading at the 18-month follow-up to stable minimal disease activity with PASI 75 and joint remission (PASI 1.2, DAPSA 2) [,] and a dramatic improvement in HRQoL (). |
A 58 year old man was diagnosed as a case of carcinoma of larynx with bilateral neck nodes (Stage T4a N2c M0; AJCC 7th Edition). As it was locally advanced case, the patient was planned for radical chemoradiation with RT doses of 76 Gy in 30 fractions over 7 weeks along with weekly chemotherapy (Cisplatin 40 mg/m2). During the course of Radiation, the patient was monitored twice a week and symptomatic care was done for RT induced toxicities. The patient presented with Grade III RD after 60 Gy in 29 fractions over 4 weeks []. The RD involved the anterior and bilateral neck with moist desquamation of the skin []. It was associated with moderate pain, difficulty in swallowing, salivation and oral mucositis grade - 2. The Patient was subsequently admitted and further treatment was initiated. Analgesics like injection Morphine 2 mg tds was administered for reducing pain and adequate hydration and nutritional support were maintained. The wound score assessed by Bates – Jensen wound assessment tool and which was 47 which falls in extreme severity status.\nIn view of extreme severe status of the RD, after cleaning of the wound with Neem water and then applied honey. The wound cleaning was regularly done, two times a day. There was a gradual improvement in RD [] at day-6th. By day 12th, the wound was healed significantly [] and day 18th wound got almost complete recovery []. The status of wound score and pain score (Visual analog score) is tabulated in . Radiation therapy was withheld for 6 days and was resumed after the improvement of RD on day – 6 []. The patient was re-started on RT and he completed his scheduled RT doses of 76 Gy in 30 fractions over 7 weeks. We here in describe a case report of locally advanced carcinoma of larynx that developed grade III RD while receiving radical chemoradiation. The patient was effectively managed with Neem (Azadirachta Indica) and Honey home remedies with remarkable results. This brief report highlights the importance of raising clinical awareness on management of RD with Neem (Azadirachta Indica) and honey. |
A 12-year-old female patient weighing 35kg was admitted to hospital with complaints of behavioral disturbances since last six weeks. Her past medical history revealed that 14 weeks ago she was hospitalized following three episodes of seizures in a day with up rolling of eyeballs, confusion and limbs stiffness. A diagnosis of complex partial seizure with secondary generalized seizure disorder was made. She was treated with tablet valproic acid 200 mg twice daily and tablet clobazam 5mg at night during her hospital stay. After 5 days of treatment she was discharged with refilled prescription of valproic acid and clobazam and advised for monthly visits. Patient was otherwise normal and had no known psychiatric history or previous episodes of abnormal behavior.\nAfter 13 weeks of treatment with valproic acid and clobazam, she was hospitalized with complaints of irrelevant talk, talking to self, wandering and singing devotional songs, decreased food intake and insomnia since about five weeks i.e. about eight weeks after initiation of valproic acid therapy. Upon hospitalization, tablet chlorpromazine 100mg once daily, tablet olanzapine 5mg thrice daily, tablet risperidone 3mg twice daily were started while she continued to receive valproic acid and clobazam at the same dose. She was discharged on day three of hospitalization with an advice to continue all medications prescribed during the hospital stay at same doses for 15 days. However, her complaints of wandering, insomnia and decreased food intake continued to persist and she was readmitted to the hospital after a week.\nDuring the present hospital admission patient was under observation and continued to receive valproic acid and clobazam. The antipsychotics were discontinued. As there was no improvement in behavior, valproic acid was suspected as a causative agent for abnormal behavior and was replaced with tablet carbamazepine 200 mg twice daily on day six of her hospital stay. Three days after the withdrawal of valproic acid her sleep pattern, food intake and speech improved significantly. A week later she was discharged with carbamazepine and clobazam with an advice for the review after two weeks. She recovered completely after 12 weeks. Later she continued to receive only carbamazepine for her seizure disorder. |
A 65-year-old Japanese man was hospitalized at another general hospital to undergo a bypass surgery for left middle cerebral artery stenosis. A few weeks before hospitalization, he had an ulcer on the oral mucosa, which sometimes oozed with blood; however, hemostasis was achieved spontaneously. After hospitalization, large hematomas with continuous hemorrhage developed in his oral cavity; therefore, he was transferred to our hospital for hematomas management. According to the physicians at the previous hospital, the patient had a history of hypertension and diabetes mellitus with chronic kidney disease (not requiring dialysis), and his activated partial thromboplastin time (aPTT) had been increasing over the past 3 months.\nOn examination, his consciousness was clear, and his vital signs were within the normal limits. No purpura was observed on the entire skin surface. His face had no swellings, and no other significant findings were observed; however, he could not close his mouth because of the presence of two large intraoral hematomas that occupied the oral cavity. The hematomas were approximately 60 (right buccal mucosa) and 30 mm in diameter (left buccal mucosa) (Fig. ). Laboratory tests revealed that his anemia had become more advanced since his admission to the previous hospital. He had a significantly prolonged aPTT of 84.8 seconds, a red blood cell count of 230 × 104/µl, hemoglobin level of 7.4 g/dl, and hematocrit level of 21.7%. A computed tomography (CT) angiogram revealed no blood vessels flowing in or out of the hematomas, and there were no findings of arteriovenous malformations (Fig. ). No obstructions or deviations were observed in the upper airway. After physical examination, coagulopathy was suspected, and an emergency surgical hemostasis was planned because of the risk of hematomas rupture and severe hemorrhage.\nWe resected the intraoral hematomas and achieved surgical hemostasis under general anesthesia and nasal intubation with fiberoptic bronchoscopy. When the hematomas on both sides of the buccal mucosa were removed by careful blunt dissection, oozing of blood was observed and hemostasis was completed by cauterization. The wound in the right buccal mucosa was replaced by an artificial dermis, and that on the left was closed by reefing. The excised hematomas were sent for histological examination. The pathological findings showed no evidence of tumor in the hematomas (Fig. ).\nPostoperatively, the patient was admitted to the surgical intensive care unit with intubation, because of the possibility of epistaxis on removal of the endotracheal tube. Further, postoperatively, a few new hematomas < 10 mm in diameter arose on both sides of the velum at a different site from the surgical site (Fig. ). These hematomas were far from the patient’s teeth and tongue; therefore, they were left in place. Although epistaxis was observed, after consulting with the hematologist and otolaryngologist, the nasotracheal tube was removed after administration of recombinant activated factor VII (rFVIIa) because AHA was suspected. Thereafter, fiberoptic examination was performed through the airway to confirm the absence of hemorrhage in the upper airway. Cross-mixing test showed a pattern of deficiency rather than a typical pattern of a coagulation factor inhibitor; therefore, initiation of immunosuppressive treatment was withheld. A few days after the surgery, purpura in the right upper and lower extremities that was not observed preoperatively became visible (Fig. ). Blood transfusion and administration of rFVIIa were performed when purpura was observed or when the hemoglobin levels were < 6.0 g/dl. Abdominal CT and laboratory tests showed no evidence of malignancy or autoimmune disease. The dose of the factor VIII inhibitor was 165 Bethesda units at 7 days after surgery, following which a definitive diagnosis of AHA was made. The immunosuppressive treatment for eradication of the inhibitor was initiated with 55 mg/day of orally administered prednisolone. Gradually, anemia improved and the inhibitor level decreased (Fig. ). The patient was discharged from the hospital on day 46. Oral administration of prednisolone 45 mg/day and follow-up examination were suggested. |
A 46-year-old Asian woman was referred to our department for a renal angiogram following 8 months of uncontrolled hypertension despite receiving medications. Initially, the patient presented with severe headache and fatigue. She had no history of smoking or drinking alcohol, was not diabetic, and had no history of diabetes in her family. She had no history of atherosclerosis. Apart from high blood pressure, the result of her physical examination was unremarkable; her general, cardiovascular system, respiratory system, and abdominal examinations were unremarkable. Neurological examination on admission showed that the patient was alert, attentive, and oriented. Her speech was clear and fluent with good repetition, comprehension, and naming. She recalled 3/3 objects at 5 min. All of her cranial nerves were intact. Motor examination revealed no pronator drift of outstretched arms. Her muscle bulk and tone were normal. Her strength was full bilaterally. Her reflexes and sensory were both intact. Her coordination and gait were normal. Laboratory investigations revealed normal complete blood count, serum cholesterol, lipid profile, and renal function (serum creatinine 119 μmol/L). Her left kidney size was normal with measurement of 9.6 cm by 4.8 cm. Renal Doppler ultrasound confirmed renal artery stenosis with renal resistive index of 0.58. The percentage of renal artery stenosis in the two branches of the left renal artery was 70% and 75%, respectively , before the first balloon angioplasty; after the first balloon angioplasty, these percentages remained the same. After the second ballooning and stenting procedure, revascularization was achieved. The patient had been attending a hypertension clinic and receiving antihypertensive drugs for the past 8 months on a regular basis under close observation. Despite this treatment and care, her blood pressure remained high at 175/110 mmHg, which the attending doctor concluded to be uncontrolled blood pressure. Initial imaging indicated left renal artery stenosis, and the patient was referred to our department (Fig. ). Prior to the diagnosis of renal artery stenosis, the patient had been receiving amlodipine 10 mg twice daily, bisoprolol 10 mg twice daily, and indapamide 2.5 mg every morning.\nThe procedure was performed under the guidance of digital subtraction angiography (floor-mounted Artis zee; Siemens Medical Solutions, Munich, Germany) using the Seldinger technique. With the patient under local anesthesia, the right femoral artery was punctured by a 21-gauge vascular access needle with an angled tip 0.035-inch guidewire, then catheterized with a 5-French introducer sheath (Terumo Interventional Systems, Tokyo, Japan). The first aortogram was obtained using a pigtail catheter (Fig. a), then an 8-French guiding catheter (Cook Medical, Bloomington, IN, USA; Cordis, Hialeah, FL, USA) was used to obtain selective renal angiograms whereby the proximal main flow and the stenosis of both branches and their respective distal flow on the left renal side were revealed. The right renal artery was normal in appearance. The left renal artery angiogram then was used as a reference for further guided interventional procedures in which the individual length and diameter of stenosis were measured. The decision was reached to perform percutaneous transluminal renal angioplasty, and the length and diameter of balloon needed were calibrated. With two balloons of 4 mm × 18 mm (Biotronik, Berlin, Germany), both were was dilated at the same time. Despite expert effort in dilatation, the stenosis was observed to persist (Fig. c). Stent placement was considered, and the procedure was continued. A preprocedure intravenous bolus of 5000 IU of heparin was administered. By using two 0.014-inch guidewires (V14; Boston Scientific, Natick, MA, USA), the interventional radiologist guided the stent to cross the upper and lower branches, respectively, through the same vascular sheath (Fig. b). Two balloon expandable stents measuring 4 mm × 18 mm and 5 mm × 18 mm (Biotronik) were placed in parallel (kissing) and simultaneously inflated both branches. A good angiographic result was revealed (Fig. d) with no need for further ballooning. Angiography contrast media (Omnipaque 350; GE Healthcare, Shanghai, China) were used. Volumes of 25 ml of contrast agent were injected at a flow rate of 5 ml/s. The final angiogram was obtained to confirm the position of the stent, the patency of the lumen, and distal blood flow. Finally, the femoral access site was closed with Perclose ProGlide (Abbott Vascular, Chicago, IL, USA). After the procedure, the patient was admitted in the ambulatory room for further observation. Her blood pressure was monitored and recorded, it showed a significant reduction of blood pressure to 128/87 mmHg. After 24 h of observation, the patient was discharged to home with aspirin (100 mg/day) and clopidogrel (75 mg/day for 3 months). During 12 months of follow-up, the patient remained well with blood pressure of 126/87 mmHg. Renal ultrasound showed bilateral kidneys of normal size and shape with good cortical medullary differentiation. A bilateral renal Doppler study appeared normal. |
A 65-year-old woman was referred to a neurosurgeon for a consultation because of a recent decrease in visual acuity (VA), which leads to the discovery of a giant olfactory groove meningioma with compression of the optic chiasm [Figures and and ]. The patient complained of a one-year history of asthenia and problems with attention. An ophthalmic examination revealed a decrease in VA in the right eye (20/50) and left eye (20/40). Dilated fundus examination and color fundus examination showed a normal optic disc in both eyes, i.e., no edema or atrophy of the optic disc. No routine color vision test was performed to complete the assessment of the optic nerve function.\nInitial visual field testing showed an incomplete temporal hemianopsia and a bilateral blind spot enlargement []. Magnetic resonance imaging (MRI) confirmed our suspicion of an olfactory groove meningioma measuring 69 cm3 and a sphenoidal planum meningioma measuring 69 cm3 was identified. She had been treated with CA for hyperandrogenism for 15 years. The treatment was immediately discontinued.\nOphthalmic monitoring and repeat MRI were scheduled, with planning for surgical intervention in case of further visual decline After administration of a steroidal anti-inflammatory drug for 15 days, repeat ophthalmic examination revealed a dramatic improvement in the patient's visual function with normalization of VA in both eyes (20/20) and improvement in her visual fields []. A MRI performed 5 weeks after cessation of CA showed a slight reduction in tumor volume from 69 cm3 to 65 cm3.\nIn view of the favorable initial clinical evolution, the observation period was extended with close imaging and ophthalmic monitoring. The patient's VA remained normal and a subsequent improvement in her visual fields was documented []. Optical coherence tomography was repeated 3 months after the first examination and showed a slight bilateral decrease in temporal retinal nerve fiber layer thickness. This decrease remained stable one year after the first examination. The initial neurocognitive symptoms also regressed significantly and the patient's work skills improved.\nMRI performed 5 and 13 months after cessation of CA confirmed a clear and long-lasting reduction in tumor volume, with a decrease from 69 cm3 at baseline to 35 cm3 on the final MRI performed at 13 months [Figures and and ]. |
A 61-year-old male patient presented to our emergency with a 15-day history of constipation and multiple episodes of nonbilious vomiting. He had no surgical history, no comorbidities, and denied any abdominal trauma in the past. The patient was evaluated elsewhere and was given a trial of conservative management. He had undergone a contrast-enhanced computed tomography (CECT) scan of abdomen and pelvis that revealed 1.5 L of ascites which was hemorrhagic on tap. The patient was conscious, oriented, and afebrile on admission with a pulse of 100 beats per minute and blood pressure of 120/86 mm Hg. Physical examination revealed a soft but distended abdomen that was not tender on palpation. Per rectal examination revealed an empty rectum and no stains on returning finger.\nAll laboratory parameters were within normal limits. No air fluid levels were seen on an abdominal X-ray in erect position. A repeat CECT of abdomen and pelvis revealed no signs of intestinal obstruction, ischemic bowel disease, ascites, or malignancy. However, compression of SMV was seen (\n). A nasogastric tube was inserted that drained 700 mL of bilious output within 1 hour. In view of the above findings, decision was taken to explore the patient.\nWe observed 15 cm of dusky bowel herniating through a 10 cm × 6 cm mesenteric defect in the terminal ileum (\n). The entire bowel in the SMV territory was dusky in appearance. The edge of the defect was fibrotic indicating a long-standing nature of the defect. No adhesions were seen near the defect (\n). No blood or free fluid was present in the peritoneal cavity. On reduction in the herniated bowel loop, complete reversal of the bowel color was noted. Viability of the bowel loops was confirmed and the mesenteric defect was closed with 3–0 nonabsorbable interrupted sutures (\n).\nThe postoperative course of the patient was uneventful. Patient was gradually weaned on to full diet by postoperative day 4 and was discharged 8 days after the surgery. |
We present the case of a 51-year-old woman with a history of Guillain-Barré syndrome as a teenager, probable neuroborreliosis in the 1980s, and a malignant melanoma in situ in the 1990s. She has given informed consent to this report. The patient had repeat rectal surgeries for sphincter damage after childbirth, and in 2011, she was diagnosed with a T4 rectal cancer causing haemorrhage. No distant metastases were found and treatment with curative intent was decided at a multidisciplinary team conference. She had neoadjuvant chemotherapy with fluorouracil and leucovorin followed by radiation with 50.4 Gy to the rectum. The tumour was resected half a year after the diagnosis. The postoperative course was uneventful except for a superficial wound infection and she was discharged after 3 weeks of hospitalization. After 2 weeks, she was readmitted because of a Takotsubo cardiomyopathy and intestinal obstruction requiring more surgery with resection of a short segment of small bowel. After 1 year, the first lung metastases were diagnosed with a 2-cm metastasis in the right lower lobe and a 5-mm. tumour in the lower left lobe. After 3 pulmonary resective procedures, further lung metastases were seen during follow-up. A second line of chemotherapy was started with oxaliplatin, resulting in shrinkage of the noted lesions. Then, local treatment was done with SBRT, 17 Gy ×3 for 2 right-sided metastases and a resection of the left lung, 3 years after the resection of the primary tumour. Three months later a new lung metastasis was diagnosed and treated with SBRT, 7 Gy ×8. Six months later, 2 more lung lesions were found and a third line of chemotherapy based on irinotecan was started, but as tumour progression was observed, treatment was discontinued after 3 months.\nWithout having the final test result on microsatellite instability in the tumour, it was decided to start PD-1 treatment (this was not within a clinical trial, since the only available trial in Sweden had not opened yet). Further resections were not possible and the given dose of radiation precludes further SBRT. There were a total of 6 confirmed metastases on both sides after the first 3 doses of pembrolizumab (standard dose with 2 mg/kg every third week). The CT evaluation showed size increase of all lung metastases, but no new lesions. The only side effect was fatigue. This early evaluation did not contradict further therapy, but before continuing with the immunological treatment, local ablative treatment with microwaves was proposed, as this has become a standard treatment for liver tumours, and also evidenced with a growing pile of international publications on treatment of lung lesions [, , , ]. The tumours were deemed treatable and were, after 2 more doses of pembrolizumab, done so in 2 sessions 3 weeks apart using a computer-assisted targeting system (CAS-one; Cascination AG, Bern, Switzerland) and microwaves (Angiodynamics) under immobilization of the lungs with high-flow jet ventilation as described in a previous publication []. The patient stayed in hospital overnight after the ablations. During the first ablative session, a pneumothorax was caused and a 16F chest tube with suction was placed and removed after clamping the next day. Images of the procedures are shown in Figure .\nPD-1 treatment was re-started after 2 weeks. After the sixth course of pembrolizumab, the patient felt pain over the liver area, and experienced low appetite and some weight loss. Further investigations showed no signs of liver metastases or damage, but a drop in serum albumin and elevated ESR and CRP. This rise in inflammatory parameters was judged as potential reaction on PD-1 inhibition and treated with betamethasone. For better pain control, amitriptyline was added to the standard analgesic regimen as there could be a neurogenic pain component. After 3 weeks, the seventh dose of pembrolizumab could be given. Two weeks thereafter, the patient experienced weakness of her left lower leg, finger tremors, and numbness on the ulnar side of the right hand. A neurologist ruled out any suspicion of new metastases or any systemic neurological disease. The symptoms were regarded as side effects of pembrolizumab, although mechanistically not fully understood. The dose of corticosteroids was increased and the eighth course of pembrolizumab was given with no worsening of symptoms.\nImages from a follow-up computed tomography are shown in Figure where an inflammatory reaction is initially seen with focal lesions, pseudo-tumours, that later disappeared. After 8 months of follow-up, there have been no signs of new or recurrent lung metastases.\nPresently, the patient is still suffering from neurological symptoms that in spite of efforts giving higher doses of steroids are not declining. The other complication is upcoming diffuse non-malignant lesions in both lungs, interpreted as either inflammatory or infectious. These findings were accompanied by shortness of breath and difficulties to walk longer distances. Steroid treatment has only partially alleviated the symptoms, as has antibiotic treatment, covering both gram-positive bacteria and Pneumocystis carinii. Further investigations are ongoing. As mentioned, the lung metastasis has not recurred, giving her time off anti-tumoural treatment. |
A 76-year-old woman presented to our hospital with a mass occurring on the skin of her right chest wall. She had been diagnosed with right breast cancer (T1N0M0, stage I) 9 years previously and had received breast-conserving surgery, sentinel lymph node biopsy, and adjuvant chemotherapy and radiation therapy for the residual whole right breast at a previous hospital. She then developed pigmented skin on her right breast 6 years after surgery, and this lesion was diagnosed as an angiosarcoma. The patient underwent a breast mastectomy to treat for RAAS. Following this, however, the angiosarcoma on her chest wall recurred three times within 2 years. The angiosarcoma was resected each time, and she received radiation therapy to her chest wall after the third operation. Four years after the first occurrence of RAAS, we observed light pigmentation and a dark red tumor (gross diameter of 5 mm) on her right chest wall (Fig. ). Clinically, recurrence of RAAS was suspected, and recurrence of angiosarcoma was diagnosed by biopsy. We considered that it was necessary to remove the irradiated skin as much as possible in order to cure the RAAS. After extensive resection of the irradiated skin and tumor, new skin collected from her right thigh was grafted to the site (Fig. ). Pathologically, the tumor size was 6 mm and the surgical margin was negative. Histologically, there were many spindle cells and dilated vascular channels. Immunostaining showed that the tumor was CD31-positive and mildly positive for CD34 (Fig. ). Ki-67 index was also high. It was revealed that there is no inconsistency as recurrence of RAAS is pathological. After the operation, the patient was hospitalized for 30 days and did not experience any complications. Although some reports suggest chemotherapy can be used to treat RAAS, we considered that this option would offer little benefit in this case, because the patient was elderly and had a history of cerebral infarction. Indeed, the patient has remained angiosarcoma-free for the last 3 years following our intervention, even without chemotherapy (Fig. ). |
A 32-yr-old woman was referred to our institution for the evaluation of an oropharyngeal mass. She reported that the mass was found incidentally at the age of three. However, because it was asymptomatic there was no further diagnostic work up or treatment. The patient had no symptoms such as swallowing difficulties or upper airway obstruction; however, she did report an occasional foreign body sensation. Flexible laryngoscopy revealed a pedunculated mass attached to the left posterior oropharyngeal wall (). A Gadolinium-enhanced T1 weighted MRI showed a 5×3×1.5 cm tumor that originated from the posterior pharyngeal wall with central enhancement and hypointense portions (). The mass had not invaded the adjoining structures. The peduncle of the mass was located at the level of C3 and the inferior portion of the mass filled the left pyriform sinus. These radiologic findings suggested a rare benign pharyngeal tumor such as a fibrous polyp and rhabdomyoma, but the radiologic diagnosis was not clear.\nAlthough the mass didn't cause any symptoms at that time and it showed completely benign features according to the history and the MRI finding, we recommended a simple excision since the mass had the potential risk of upper airway obstruction due to its location just above the larynx and its huge size. A preoperative procedure for making the histological diagnosis was not performed because of the potential risk of blood aspiration and airway obstruction caused by swelling that would occur after the biopsy at the out-patient department.\nUnder general anesthesia, a pedunculated 5×3×1.5 cm sized mass was exposed with using a McIvor mouth retractor. The mass was completely excised from the posterior wall of the oropharynx with using monopolar electrocautery. The prevertebral fascia was exposed after the resection; the mass had not infiltrated to the adjacent structures. Frozen biopsy was not performed since the mass had benign features and an adequate resection margin was acquired. The patient was discharged on the same day of the operation. At three months follow up, the wound had healed completely and there was no evidence of recurrence or a residual mass.\nGross examination of the specimen showed a firm mass with a homogeneous cut surface. Histopathological examination of the specimen revealed that some foci of the lesion were lined by heterotopic gastric foveolar epithelium with gastric glands beneath the surface. In addition, there were glandular structures that were characterized by parietal and chief cells. Moreover, there was focal intestinal metaplasia that contained goblet cells in the glands. The muscularis propria was found beneath the submucosal layer. These findings were consistent with gastric choristoma (). |
A 45-year-old male was admitted to the emergency department with complaints of acute onset severe abdominal pain and vomiting for three days. He was earlier evaluated and treated at a local hospital where he was kept on conservative management with insertion of nasogastric tube for 24 hours. But his symptoms were further aggravated with increased bouts of vomiting; as a result he was referred to our institute for further management. The patient had no history of similar attack in the past. He did not report any fever, dysuria, and change in bowel habit or hematochezia. There was no history of any comorbid illness, past surgical intervention, or trauma. On general examination, pulse rate was 98/minute and blood pressure was 130/80 mm of Hg. Patient was afebrile and tachypneic. Abdominal distension and tenderness were detected on abdominal examination. Bowel sounds were decreased. Digital rectal examination was normal. Rest systemic examination revealed no abnormality. Abdominal radiography in the upright position showed dilated small intestinal loops with multiple air-fluid levels suggesting intestinal obstruction. Ultrasonography of the abdomen revealed gaseous distension of the bowel loops. Routine blood investigations were within normal limit.\nThe patient was planned for exploratory laparotomy in view of acute intestinal obstruction. On laparotomy, a loop of ileum was found to enter the retroperitoneum through a hernia gate (defect of 2.5 cm) located lateral to the sigmoid colon in the left paracolic gutter (Figures and ). The small intestinal loop was incarcerated at the neck of the hernia sac which was reduced after widening the hernia gate. The segment of intestine was found to be viable after reduction for which resection was not required. The hernia gate was closed with nonabsorbable interrupted suturing (). There was no malrotation of the gut. Postoperative recovery was uneventful and the patient was discharged on the sixth postoperative day. He is now on follow-up and is doing well for the past six months after surgery without any symptoms. |
A 38-year-old male presented to the surgery outpatient department with complaints of odynophagia and chest pain for two weeks. The symptoms had started after a meal when the patient felt the sensation of a foreign body in the throat, which he reportedly swallowed with a bolus of food. As the initial evaluation with neck and chest X-rays was reported as normal, he did not receive any specific intervention. However, in the third week after ingestion, he developed chest pain and fever. On evaluation with a contrast-enhanced thorax, chest, and abdomen scan, he was diagnosed with a right-sided pleural effusion and a suspected foreign body in the periesophageal region of the lower thoracic esophagus close to the inferior vena cava (Figures , ).\nFrom the hiatus region, the foreign body was located at approximately 5 cm cephalad. An intercostal drain was placed under image guidance and approximately 750 mL of serosanguinous pleural effusion was drained. An upper gastrointestinal contrast study did not show any contrast extravasation from the esophageal lumen. Upper gastrointestinal endoscopy also did not show any signs of recent perforation or a foreign body in the esophagus. An after adequate chest optimization, he underwent laparoscopic foreign body removal.\nThe procedure was done under general anesthesia in the supine split leg position. After initial access and the creation of a pneumoperitoneum through the left paramedian 12 mm port using an open method, five additional 5 mm ports were placed, including two midclavicular line ports on either side, a left subcostal port for assistance, and an epigastric port for liver retraction. The gastrohepatic ligament was divided to reach the right crus. The phrenoesophageal membrane was then opened to enter the mediastinum. The esophagus was then looped with an umbilical tape at the level of the hiatus and used for subsequent retraction. The right side of the esophagus was dissected meticulously, preserving the vagus nerves. At about 6 cm from the gastro esophageal junction, there was a fibrotic tract along the lateral aspect, which was then cut with scissors exposing the 2.7 cm long metallic foreign body (Figure ).\nRight pleura were thickened and were not opened. The foreign body was then extracted and removed under vision (Figure ).\nThe tract, which was seen extending to the esophagus, was clipped on the esophageal side using a hemolock. A thorough mediastinal lavage was given and a 16 Fr suction drain was placed in the paraesophageal region. An upper gastrointestinal oral contrast study that was done on the second postoperative day did not reveal any contrast leak, and the patient was started on an oral diet. He had an uneventful postoperative course and was discharged on the fifth postoperative day. |
A 6-year-old boy with no pathological history accidentally fell from the top of an approximately 3 m climbing pole and injured his right extended elbow and wrist joint. Due to pain and deformity in the right elbow and wrist joints, he visited our hospital. Swelling and a dinner fork deformity of the right wrist joint and pronounced swelling of the right elbow joint were observed. No skin damage was observed. No findings of nerve injury or arterial injury were obtained in the right upper limb. Radiography revealed lateral dislocation of the radial head, a fracture of the proximal ulnar metaphysis, and mild bending deformation at the fracture site. In addition, fractures of the distal radius and ulna, as well as dorsal displacement of the distal fragment, were seen (). Thus, the patient was diagnosed with Bado type III Monteggia injury with ipsilateral fracture of the distal radius and ulna.\nManual reduction under nerve block was attempted on the day of injury. However, because it was difficult to maintain the reduction of the radial head, as shown in , open reduction and percutaneous procedures were performed under general anesthesia. A Kirschner wire was inserted, percutaneously, from the olecranon into the ulnar diaphysis. When the Kirschner wire was in place, the dislocation of the radial head immediately showed good reduction. Further, open reduction and fixation of the fractured distal radius and ulna were performed with Kirschner wires (). A long-arm cast was used for external fixation with the elbow in 90° flexion and the forearm in an intermediate position.\nTwo weeks after surgery, callus formation at the fractured bone was observed. Therefore, the cast was removed, and range of motion (ROM) exercises of the elbow and wrist joints were initiated. Since bone union was achieved at 6 weeks postsurgery, the Kirschner wires were removed. Pain, ROM limitation, and lateral instability were not observed in the elbow or wrist joints at 3 months after surgery. Additionally, plain radiographs taken at the same time showed a radially convex curvature at the proximal portion of the ulna and lateral subluxation of the radial head (). However, a gradual correction in the outward displacement of the radial head was observed during the 3-year follow-up.\nTwenty-one years after surgery, the patient returned to our hospital for another disorder. At that time, we obtained informed consent to perform an examination and take radiographs of the previous Monteggia injury. Neither spontaneous pain, pain during exercise, tenderness, nor ROM asymmetry were observed (). The biocompatibility of the radiocapitellar joint was good, and no malunion was found in the distal radius and ulna (). The patient reports that he has been working as a computer programmer and performs weight training as a hobby without limitations. |
A 62-year-old woman (Figure , II-3) was admitted to the hospital for recurrent partial headache with weakness of one side and aphasia for about 45 years. In her first attack, the patient suddenly experienced an aura with visual disturbances which she described as increasing scotomata in the bilateral visual field. After a few seconds, the patient developed a serious headache, mainly located on the left side. After a few minutes, she presented a paralysis of the right side and speech difficulties accompanied by dizziness and vomiting. These symptoms resolved after about 2 h. After this initial onset, she had an attack nearly every 4–5 years, and the clinical presentations of her attacks were always similar to the first one. The duration of the aura symptoms and the migraine was typically 1–2 h but sometimes the migraine could last up to 4 days. Sometimes headaches occurred before the hemiplegia and aphasia. Each headache was accompanied by dizziness and vomiting but without loss of consciousness. In most attacks, this patient experienced additionally a flushing of the neck and face and felt that the skin temperature of this affected area was increased, but the temperature was never measured. These symptoms may be related to an extracranial vasodilation when a migraine attack occurred. She did not undergo regular treatment except for simple analgesics as a symptomatic therapy. Recently, her condition aggravated as the frequency of attacks increased from once every 4–5 years to once every 1–2 weeks which had a serious impact on her everyday life. Therefore, during a severe migraine attack, she visited our hospital. We reviewed her family history, and we found that three other subjects, her mother, brother, and nephew, had similar clinical symptoms (Figure ). Their presentations are as follows:\nThe proband's mother (Figure , I-2) died of uremia at the age of 72. According to her husband and children, she reported typical hemiplegic migraines since an age of 14 years with five attacks per year on average. The aura symptoms were similar to those in the proband, including bilateral visual symptoms (scotomata), speech difficulties, and hemiparesis.\nThe proband's 55-year-old brother (Figure , II-1) had first at the age of 15 headache attacks with nausea, vomiting, visual field defects, and one-sided motor weakness. Usually, these attacks last 5 h. The disease presentation was progressive with age.\nThe proband's nephew (Figure , III-1), a 25-year-old fitness coach, had first headache attacks with visual symptoms (scotomata) and lateralized motor weakness at the age of 13. Each attack lasts about 20 min.\nAfter admission, her neurological examinations were unremarkable and brain magnetic resonance imaging (MRI) and Magnetic Resonance Angiography (MRA) showed no meaningful abnormalities (Figure ). Thus, the suspected diagnosis was transient ischemic attack (TIA). During her hospitalization, the patient had several migraine attacks that were characterized first by visual symptoms, then aphasia and right limb paralysis 10 min later, and finally severe headaches after 20 min. At that time, the neurological examination revealed: no loss of consciousness, motor aphasia, muscle strength 2 in the right limb, and normal findings in the examination of the residual nervous system. After about 1 h, the symptoms of the aura were relieved, while the headache lasted for about 1 day. However, the symptoms were not relieved after dual antiplatelet aggregation treatment, and transthoracic echocardiography and carotid ultrasound failed to identify any underlying cerebrovascular etiology. After careful consideration of all aspects, she was diagnosed with hemiplegic migraine. So, we conducted a genetic test on the patient and found a heterozygous point mutation (c.4495T>C) in exon 26 of the SCN1A gene. This mutation caused amino acid 1499 to change from phenylalanine to leucine (p. Phe1499Leu), which may cause the disease by affecting the SCN1A protein function.\nTo establish the diagnosis, we performed a genetic test on those family members to analyze for the presence of mutations in genes including CACNA1A, ATP1A2, and SCN1A related to FHM. We only found a gene mutation in SCN1A, but this mutation was detected in all affected subjects in this family (Figure ). Therefore, this patient was diagnosed with FHM3. She was discharged after receiving a health education on migraine attacks, which suggested her staying away from stress, bright lights, sleep disturbances, physical exertion, and alcohol consumption because these have all been reported as trigger factors in FHM (). Upon being discharged from the hospital, she had intermittently taken flunarizine capsules and rizatriptan benzoate tablets to prevent and control migraine attacks. After 6 months of follow-up, the efficacy of the drug was uncertain, because the frequency of headache attacks was not adequately reduced. After the low efficacy of her medication became clear, we consulted again the literature and consider now a trial with lamotrigine or acetazolamide (). |
A 76-year-old woman presented to our hospital with a mass occurring on the skin of her right chest wall. She had been diagnosed with right breast cancer (T1N0M0, stage I) 9 years previously and had received breast-conserving surgery, sentinel lymph node biopsy, and adjuvant chemotherapy and radiation therapy for the residual whole right breast at a previous hospital. She then developed pigmented skin on her right breast 6 years after surgery, and this lesion was diagnosed as an angiosarcoma. The patient underwent a breast mastectomy to treat for RAAS. Following this, however, the angiosarcoma on her chest wall recurred three times within 2 years. The angiosarcoma was resected each time, and she received radiation therapy to her chest wall after the third operation. Four years after the first occurrence of RAAS, we observed light pigmentation and a dark red tumor (gross diameter of 5 mm) on her right chest wall (Fig. ). Clinically, recurrence of RAAS was suspected, and recurrence of angiosarcoma was diagnosed by biopsy. We considered that it was necessary to remove the irradiated skin as much as possible in order to cure the RAAS. After extensive resection of the irradiated skin and tumor, new skin collected from her right thigh was grafted to the site (Fig. ). Pathologically, the tumor size was 6 mm and the surgical margin was negative. Histologically, there were many spindle cells and dilated vascular channels. Immunostaining showed that the tumor was CD31-positive and mildly positive for CD34 (Fig. ). Ki-67 index was also high. It was revealed that there is no inconsistency as recurrence of RAAS is pathological. After the operation, the patient was hospitalized for 30 days and did not experience any complications. Although some reports suggest chemotherapy can be used to treat RAAS, we considered that this option would offer little benefit in this case, because the patient was elderly and had a history of cerebral infarction. Indeed, the patient has remained angiosarcoma-free for the last 3 years following our intervention, even without chemotherapy (Fig. ). |
A previously healthy 42 years old male presented to our institute with history of gradually progressive and painless swelling over left calf since two months. He was a non-smoker, laborer by occupation. The patient noticed a firm swelling in calf region of left leg 5 years ago. No history of trauma or infection prior to the appearance of the mass was reported. No family history of any such swelling in the past. Patient was operated for swelling over calf region 20 years back but no records were available. There was a history of gradual increase in deformity of left foot since 3 years and the patient had started walking on toes on left side with no dorsiflexion at ankle joint.\nOn examination, there was a single, non-tender, hyperpigmented scar of size 8×3 cm over mid-calf region fixed to underlying structures. A large, well defined non-tender, firm, swelling was palpable in posterior aspect of left leg measuring about 28×8 cm extending from tendoachilles region up to 5 cm distal to popliteal fossa and medially and laterally up to border of tibia and fibula respectively. The overlying skin was normal with no discoloration and local raise of temperature. Movement of knee joint was normal. There was fixed equinus deformity of left foot (). No inflammatory signs, skin changes or adenopathies were present. No bruits were heard on auscultation. Neurovascular examination of left leg and foot was normal. Laboratory findings were within normal limits. Radiological examination revealed large soft tissue mass with linear and streak-like ossification around the left tibia. MR Angiography () showed arteriovenous malformation in left calf with multiple feeding arteries arising from popliteal, peroneal and anterior tibial artery and large draining veins draining deep into venous system of leg. The tibia and fibula marrow showed normal signal intensity.\nBecause of patient symptoms and with clinical diagnosis of a vascular malformation, a wide surgical excision of the lesion was done. Through a 25 cm longitudinal incision across the calf, posterior compartment muscles were exposed. The mass was found completely involving superficial group of posterior compartment muscles sparing the deep compartment with no attachment to periosteum or bone (). Peroneal artery and vein were found to be embedded in the lesion and thus sacrificed. Plane of dissection was between superficial and deep muscles.\nThe lesion was completely removed along with overlying cutaneous scar with wide surgical margins leaving posterior tibial artery in continuity. Intraoperative, complete dorsiflexion of foot was achieved with intact vascularity of leg. The excised specimen was very hard like bone and had to be cut longitudinally with saw (). Grossly the resected specimen showed ossified tissue covered with skin and soft tissues including muscle, tendons and adipose tissue measuring 15×7×5 cm. The cut surface of the ossified area was grey white, gritty and congested (). Microscopically, it revealed features of a vascular malformation with numerous blood vessels of variable size and shape composed of arteries and veins which were dissecting soft tissues and interstitial planes of skeletal muscle.\nMany of the vessels were thin walled with anastomosing and a sinusoidal appearance. Some of them showed fresh and organized thrombi within this vascular background, extensive osseous metaplasia characterized by mature lamellar bone formation was seen. (). The diagnosis was consistent with arterio-venous malformation with extensive osseous metaplasia. At the time of recent follow up after one year from the operation, no local recurrence of the tumor was demonstrated clinically and radiologically. No restriction of motion of ankle joint was found. Patient is presently walking with a normal gait. |
A 7-week-old boy was transferred to our Institution after being hospitalized for 4 weeks at another University Hospital for RSV bronchiolitis necessitating mechanical ventilation. Because the respiratory situation was not improving, he underwent flexible bronchoscopy, chest CT, and echocardiography. The examinations revealed PAS associated with a PDA, an atrial septal defect, and a diffuse LSTS with eight complete cartilaginous rings extending from the fifth tracheal ring to five millimeters above the carina (Figure ). Our Institution specializes in pediatric tracheal surgeries, therefore the surgical intervention was performed at our Institution. After doing a median sternotomy and putting the child on cardiopulmonary bypass, ligation then section of the PDA and LPA reimplantation were performed followed by slide tracheoplasty (Figures and ). Total concomitant cardiac and tracheal surgery duration was 317 minutes, of which 140 minutes were on cardiopulmonary bypass.\nRespiratory and cardiovascular problems occurred after the operation. The major respiratory problem consisted of repetitive failure of weaning from mechanical ventilation. The first attempt was done on the eighth postoperative day (POD), but the patient presented severe respiratory distress and stridor despite non-invasive ventilation and was reintubated the same day. Endoscopy revealed left vocal cord paralysis and significant bronchomalacia of both stem bronchi, without any sign of anastomotic failure at the site of the tracheoplasty. The second extubation was performed on POD 18, again followed by non-invasive ventilation. Because of severe respiratory distress, the patient was reintubated 3 days later. Endoscopy of the airways showed the development of granulation tissue at the level of the anastomosis, which was removed. On POD 28, third extubation trial was done, but 2 days later, mechanical ventilation was again required. Endoscopy revealed a constriction of the right main stem bronchus requiring balloon dilatation. The patient was successfully weaned from mechanical ventilation on POD 45, with the need for non-invasive ventilation for another 2 months. Endoscopy 3 months after the operation showed a slight stenosis of the trachea and both main stem bronchi, responding each to balloon dilatation, persisting left vocal cord paralysis and persisting but improving bilateral bronchomalacia. Despite these findings, the patient clinically improved and could be transferred to the referring hospital without any respiratory support or oxygen therapy. The last endoscopic control 1 year after the surgery showed a satisfactory postoperative result without any sign of granulation tissue or stenosis of the airway. The left vocal cord was still paralyzed with optimal right vocal cord compensation, and there was mild distal tracheal and bronchomalacia.\nFrom the cardiovascular point of view, the postoperative period was also challenging. In the context of worsening oxygenation on POD 10, echocardiography was performed showing absent LPA flow. CT scan revealed bilateral pulmonary artery thrombosis (Figure ). Therapeutic anticoagulation with unfractionated heparin was started and maintained for 2.5 weeks. Response to treatment was good, and repermeabilization of the pulmonary arteries could be demonstrated after 48 hours.\nRepeated echocardiograms showed patency of the LPA, but mild to moderate stenosis at the reimplantation site as well as discrete stenosis of the right pulmonary artery at the level of the initial origin of the LPA. Due to the persistence of these stenoses during the follow-up, percutaneous balloon dilatation was performed successfully one year after the surgery.\nThe total length of stay in hospital of this patient was 184 days, 24 days thereof preoperatively and 108 days in the pediatric intensive care unit, before he could be discharged home. The total duration of mechanical ventilation was 70 days, 25 days thereof preoperatively. On last follow-up, the patient is doing well without any respiratory symptoms. He has no bronchoaspiration while feeding, and the voice is mildly hoarse but communicable. Future follow-up appointments will assess long-term outcome and evaluate if this long hospitalization in early infancy will affect long-term neurologic development. Currently, the patient has a normal neurologic development. |
A 9-year-old female with no significant past medical or family history presented with a complaint of intermittent irregular sudden jerky movements in all the four limbs and head of 6-month duration. This abnormal movement started in the right lower limb which over the next 1 month progressed to the right upper limb, head, left lower limb, and then left upper limb. The movements used to occur asynchronously in the limbs and head lasting less than a second with no loss of consciousness, diurnal variation, clustering, and never in sleep. Because of the movements, the patient used to have occasional falls, slippage of objects from hand, and occasional spillage of food while eating. The frequency of jerks also progressed over 6 months []. For the first 3 months, these were the only symptoms. Then, 3 months from the onset, the patient started having drooling of saliva with progressive swallowing difficulty and speech problems. The symptoms progressed that at presentation the patient could eat only semisolid food with severe dysarthria and able to utter only few words. There was also inappropriate laughter. Simultaneously to the onset of bulbar symptoms, the patient also developed abnormal posturing of limbs which started in the right half of the body followed by the left with difficulty in walking and performing daily activities with hand. The posturing was associated with stiffness which used to get resolved completely in sleep. There was also poor attention with worsening performance in school. There was no history of convulsions, fever, headache, vomiting, visual complaints, motor weakness, sensory impairment, or bowel and bladder abnormalities. There was no history of jaundice, gastric complaints, bleeding tendency, or measles in the past. The patient was fully vaccinated according to the national vaccination program.\nOn examination, the patient was alert, conscious, inappropriately smiling with drooling of saliva, and persistently open mouth. Cognitive function was severely impaired. There was no auditory or visual abnormality with normal fundus examination, extraocular movement, and pupillary reflex. Kayser–Fleischer (KF) ring was evident in both eyes. There was repeated dystonic, synchronous, uprolling of both eyes without impairment in sensorium and other associated movements. Severe dysarthria and dysphagia were noted with tongue dystonia but with normal gag reflex and symmetrical palatal movement on vocalization. Motor system examination revealed generalized rigidity with normal power, normal deep tendon reflexes, and flexor plantar response. There was multifocal myoclonus involving head and all the four limbs predominately in the right half of the body. In addition, there was dystonic posturing involving all the four limbs. Sensory and cerebellar system examination was normal. On gait examination, the patient showed short shuffling gait with festination. Other system examinations were normal.\nRoutine investigation comprising complete blood count, renal function test, and liver function test was normal. Serum copper increased, serum ceruloplasmin reduced, and 24 h urine copper increased. Slit lamp examination confirmed KF ring in both eyes. Magnetic resonance imaging (MRI) of the brain revealed bilateral basal ganglia, thalamic, midbrain, and pontine hyperintensity consistent with WD []. Electroencephalogram (EEG) was normal despite multifocal myoclonus suggesting cortical origin. Ultrasonography of the abdomen did not reveal any liver abnormality.\nThe patient was diagnosed as a case of WD and started on pencillamine, zinc, and diet modification. Clonazapam was also added for myoclonus. The patient was discharged after initial observation. At 3-month follow-up, the patient had no myoclonus with improvement in extrapyramidal symptoms and static cognitive impairment. |
A 53-year-old female presented with gradually progressive diminution of vision in her left eye for 2 months. She had a history of diabetes mellitus for 20 years for which she is taking insulin. Her most recent HbA1C was 7.5%. She had no history of hypertension or renal problems. She had no past ocular history. Examination revealed a corrected distance visual acuity (CDVA) of 20/40 in her right eye and 20/100 in the left. Anterior segment examination showed nuclear sclerosis in both eyes. Posterior segment examination revealed intraretinal hemorrhages in all 4 quadrants indicating severe nonproliferative diabetic retinopathy in both eyes with clinically significant macular edema in the left eye confirmed by fluorescein angiography (). Spectral domain optical coherence tomography (OCT) of the macula was done and revealed multiple cystic spaces, mild subfoveal neurosensory detachment, and diffuse retinal thickening with a central subfield macular thickness of 332 μm in the left eye (). The right eye showed only few cystic spaces with minimal thickening. OCTA (Optovue, Inc., Fremont, CA, USA) was done in both eyes and showed areas of capillary nonperfusion in the superficial capillary plexus (SCP) of the maculae of both eyes (Figures and ). Three monthly intravitreal bevacizumab injections were done to treat the macular edema in the left eye. One month following the last intravitreal injection, CDVA improved to 20/60 in the left eye and was stable in the right eye. OCT showed improvement of the macular edema in the left eye (). OCTA was performed in both eyes and showed decreased vascular density of the SCP of the left eye compared to pretreatment OCTA while a mild increase was noted in the vascular density of the SCP of the untreated right eye (Figures and ). The patient was then followed up without requiring further intravitreal injections and 4 months following the last intravitreal injection OCTA was repeated in both eyes and showed improvement of the vascular density of the SCP of the left eye with unchanged SCP in the right eye (Figures and ). CDVA was 20/60 in the right eye and 20/100 in the left. Clinical examination revealed retinal neovascularization in the right eye with clinically significant macular edema in both eyes. OCT showed increased center-involving macular edema in both eyes with recurrent neurosensory detachment in the left eye (). Three monthly intravitreal bevacizumab injections were then done for treatment of proliferative diabetic retinopathy in the right eye and the macular edema in both eyes. One month following the last intravitreal injection, CDVA was 20/40 in the right eye and 20/60 in the left, OCT showed decreased macular thickness in both eyes (), and OCTA revealed decreased vascular density of the SCP of both eyes (Figures and ). Changes in the deep capillary plexus closely followed those in the SCP at all stages. |
A 28-year-old woman (1.65 m, 85 kg, BMI 31.2) was referred to our department five days postpartum with a bilateral femoral neck fracture.\nDuring the last two months before the delivery of her first child (begin with the sixth month of gestation), she had complained about increasing weakness and pain in both hips and thighs. In the last two weeks of her pregnancy (begin of the 35th week of gestation), she had to walk on crutches, as she was unable to bear her full weight due to the severe pain in her hips. During this time she took paracetamol daily. Her treating gynaecologist did not initiate any clarification of the symptoms by radiologic imaging methods in this period. Considering her severe pain, it was decided to deliver the child per elective caesarean section (37th +4 week of gestation). Three days postpartum, after being released from the hospital, she had an epileptic seizure and fell. She was admitted to a neurological department of another hospital. During her two-day stay in this department, the woman complained of severe pains in both hips. X-rays and a computer tomography (CT) of the pelvis were performed, revealing bilateral femoral neck fractures. Following this diagnosis, the patient was referred to our department for further treatment.\nRegarding her past medical history, she had childhood migraine and epilepsy. She suffered from her first epileptic seizure at the age of 15. She was treated with valproate over 11 years and remained seizure-free during that time. Her treating neurologist stopped the medication two years prior to her pregnancy. The patient did not have any other comorbidities. Particularly, she did not report on any past history of fractures, irregular menstruation, or family history of osteoporosis.\nAt presentation in our department, the clinical examination showed massively reduced hip movement on both sides because of severe pain. Serum laboratory examination was normal.\nBased on the age of the patient, the limited bone quality, the age of the fractures, and their displacement (Figures and ), it was decided to perform a bilateral hip joint replacement with the use of short-stemmed prostheses (). There were no intra- or postoperative complications. Both femoral heads were sent for further histopathological examination. The results of both femoral heads revealed fracture areas with hemorrhagia, proliferation of fibroblasts in the marrow cavities, and formation of focal woven bone as a sign of the bone remodelling, being indicative for a TOH. Postoperatively, the patient was mobilized on crutches under full weight bearing of the operated extremities. After consultation with our Department of Neurology, the patient restarted her valproate medication. Due to this medication, the patient was advised not to breastfeed her child. Since the TOH is regarded to be a self-limiting disorder, no specific osteoporotic treatment was applied. The further course was uneventful, and the patient was dismissed after two weeks.\nAt 1-year follow-up, the patient is free of any complaints and has no limitation in the range of motion of both hips (extension/flexion 0°-0°-100°, abduction 50°). |
A 19-year-old pregnant woman, who was a petty trader in a health facility, referred to a health facility in Freetown (Sierra Leone) with complaints about not noticing fetal movement in the last 3 days. She was pregnant for the first time and in the 36 weeks of gestation. With the clinical information of not noticing fetal movement, she was induced using oral medication and IV lines inserted for fluids. Induction occurred for several hours and she was delivered a dead baby. The placenta did not show any noticeable abnormalities but the dead child was heavily macerated. The placenta was discharged and later incinerated according to the standard clinical procedure. The safe and dignified burial team was called, swabbed the dead baby and the sample was transported to the nearest laboratory for EVD testing. The nurses who engaged the patient realized a full personal protective equipment. The surveillance team was called in to investigate the case.\nPrior to delivery, the woman clinical picture looked normal other than the in utero deceased baby as indicated by the ultra sound scan. She had no fever, no vomiting and no diarrhea. PCV and blood pressure were also normal for the gestation period. After the commencement of the induction, she experienced a small amount of vomiting, which could be related to the induction processes. After delivery, she was still normal, with no bleeding and no other issues. She was discharged home with medication (amoxicillin and flagyl). She was reported to have a history of sickle cell anemia. The placenta was discharged and then incinerated (standard procedure).\nThe pregnant woman was not known to any team member and did not come from a contact list or quarantine home and was not sought in connection with any cluster. Discussions ensued after the morning meeting, with the agreement of going to the clinic to commence investigations. The Contact Tracing Mentor telephoned to state that the woman had returned to the clinic and she has agreed to speak with the surveillance team. The woman returned because she forgot to take her discharge medications and not for any complication. The staff, knowing of the positive result of the swab, kept her at the clinic in the isolation unit. She remained in good health. She was well, alert, sitting up strongly, and speaking normally. She was symptom-free – no fever, no bleeding, no vomiting, no aches and pains etc. She did not meet the EVD case definition and she agreed to be interviewed with a hospital staff, contact tracer and WHO team. The interviewers wore personal protective equipments and maintained a distance of 1 m at all times. No materials were exchanged.\nShe did not report any travel history. She did not report any knowledge of other cases, or of having any sick people at her home. She denied having attended any funeral. The father of the child was reported to be a casual trader. The woman reported that she had not really seen him since the conception. Effort was made to contact him with a view to interviewing him to determine any similarities or contradictions with woman’s story.\nThe swab from the baby was taken to the nearest laboratory and it returned positive to direct qPCR with CT value of 21; this was again taken to another laboratory for confirmation and it also returned a positive qPCR test of 21.5 threshold. The woman was invited for sampling and a qPCR test reported negative with a CT value of 42. Her blood samples were taken for serology and returned IgM negative and IgG positive using the competitive ELISA technique which was carried out in the Chinese laboratory in Jui Freetown. |
A 53-year-old man presented with subcutaneous hydrops and abdominal distension on the 3rd postoperative day after exploratory laparotomy for intestinal obstruction due to an abdominal cocoon in another hospital. He was transferred to our department on the 7th day because of incision exudation similar to intestinal contents and progressive abdominal distension. Abdominal CT showed dehiscence of a fraction of the linea alba and subcutaneous hydrops. To verify the characteristics of subcutaneous hydrops, a skin suture was removed, and the intestinal contents flowed out of the subcutaneous cavity. When several other sutures were removed sequentially, an approximately 1-cm defect on the small intestine was identified with exposure to the air. Re-suturing of the intestinal defect and linea alba was initially attempted but failed because of the friability of the bowel wall and the severe adhesions surrounding the incision. However, severe adhesions also prevented the spread of infection. Thus, conservative treatment was the only option. Under local anesthesia, a new fistula tract measuring approximately 6 cm was constructed with embedding of the triple-cavity tube in subcutaneous tissues (Fig. ). Continuous negative pressure at −80 mm Hg was applied, and the net output exceeded 500 ml/day in the first few days. Because of the mild infection, an antibiotic was administered according to a bacterial culture sensitivity test. Octreotide and TPN were administered until controlled fistula tract formation was identified by fistulography 7 days later. After anus exhaust, an elemental diet was given, and recombinant human growth hormone was administered at a dosage of 8 U/day instead of octreotide. Oral loperamide hydrochloride capsules were administered to reduce the intestinal output. However, no apparent signs of tract growth were found when recombinant human growth hormone was administered for 3 weeks. Thus, to avoid the side effects of long-term use, recombinant human growth hormone administration was stopped. The fistula finally healed on the 48th day after tract construction. No fistula recurrence and other relevant complication appeared during follow-up at 1 week, 1 month, and 3 months.\nIn addition, clinical characteristics of all the 3 cases including fistula location, defect size, output volume, approach of triple-cavity tube placement, length of fistula tract, somatostatin and somatotropin administration time, and fistula healing time were recorded (Table ). |
A 56-year-old female with a history of uncontrolled biopsy-proven bullous pemphigoid began treatment with cyclosporine 100 mg three times a day for a total daily dose of 300 mg. After five weeks of therapy, the patient complained of nervousness and shakiness, and the cyclosporine dose was decreased to 100 mg twice daily for a total daily dose of 200 mg. After an additional five weeks of cyclosporine therapy at 200 mg daily, the patient returned with complaints of the gradual onset of redness, swelling, and pain of her left hand over the past six weeks (Figure ). On examination, erythema and violaceous swelling were present on the patient’s left hand, most predominant over the dorsal and palmar aspects of the left second and third metacarpals, and left proximal and mid phalanges. There was tenderness to palpation and pain with movement of the left hand and phalanges. The patient described the associated pain as a burning sensation. Although erythromelalgia was considered, this was believed to be a local reaction, and the cyclosporine was continued as treatment for the patient’s bullous pemphigoid.\nSeven weeks later, the patient returned complaining of significantly worsened redness, swelling, and pain of the left hand with progression of involvement of the right hand. On examination, the erythema and edema involved the dorsal and palmar aspects of the left hand and phalanges, most severely affecting the left proximal phalanges. The right hand was also visibly erythematous with edema most prominently involving the right second proximal phalanx. The patient complained of worsened pain, described as “burning similar to a sunburn” that significantly worsened upon exposure to the sun or heat. The pain also worsened with passive and active movement of the hand or digits, as well as palpation of the affected areas. The patient endorsed improvement with exposure of the hands to cold temperatures, particularly describing relief of the pain while running the hands under cold water. At this time, the cyclosporine dose was decreased to 100 mg once daily with a diagnosis of erythromelalgia secondary to cyclosporine. It was hoped that a lower dose of cyclosporine could be used to treat the bullous pemphigoid, with resolution of the symptoms of erythromelalgia.\nSix weeks later, the patient returned for a follow-up to evaluate the erythromelalgia. The erythema and edema had significantly improved, with only minor swelling of the second and third phalanges of the left hand and the second phalanx of the right hand. The patient's symptoms improved following the transition to low-dose cyclosporine therapy (100 mg once daily). At an additional follow-up eight weeks later, the patient’s symptoms of erythromelalgia had completely resolved. |
A 28-year-old woman was admitted to our hospital due to unstable gait caused by pain in the right hallux, for which she was unable to stretch for over three months.\nThe patient had undergone “resection of ameloblastoma at the right mandible, mandibulectomy, and autogenous right fibula grafting” at another hospital on November 20, 2017. The right lower limb of the patient was wrapped with gauze and fixed with a splint for over 20 d. The patient reported within 20 d that the wrapping was too tight, without any evident pain. The patient started walking with the assistance of a single crutch after the splint was removed, and could walk unaided one month after the operation. However, the hallux toe, as well as the second and third toes of the right foot could not be stretched, with pain in all the toes. Therefore, the gait of the patient was unstable, and she was susceptible to fall. Going up and downstairs was very difficult for her.\nThe patient had no significant medical history, psychiatric history, or history of substance abuse. On admission, the physical examination showed that the patient had clear consciousness, and her mental status was normal.\nA healed scar of approximately 15 cm in length was found at the jaw. Another healed scar of approximately 28 cm in length was found at the posterolateral side of the right calf. No evident swelling was found in the right calf and right foot, and no evident pressing pain was found around the right ankle. The motion of the right ankle was restricted, and the parameters were as follows: Dorsal flexion was about 0°; plantar flexion was about 45°, which was generally normal; the right hallux toe was slightly valgus; the second and third toe joints were flexed and could not be stretched when the ankle joint was in neutral position. However, these two toe joints could be stretched during plantar flexion of the ankle. The circumference of the calf was 21.5 cm and 22.5 cm at 10 cm above the right and left ankles, respectively. In addition, the circumference of the calf was 29.5 cm and 31.5 cm at 20 cm above the right and left ankles, respectively. Hypoesthesia of the skin was found at the lateral side of the right calf and right foot-back. Fluctuation of the dorsal artery was normal (Figure ).\nX-ray of the right ankle and right knee performed on March 30, 2018 (Figure ) showed discontinuity of the proximal and distal ends of the right fibula, which could be the changes following fibula osteotomy. Electromyography (EMG) performed on April 1, 2018 showed damages of the sural sensory nerves. Color ultrasound examination suggested the possibility of flexor hallucis longus injuries. Magnetic resonance imaging of the right calf on April 12, 2018 (Figure ) showed discontinuity of the bony substance of the right fibula, which was postoperative change; abnormal signal of the right flexor hallucis longus that suggested injuries; and tenosynovitis of the right flexor hallucis longus. |
A 61-year-old man with no remarkable history had never had a regular medical examination. He has been smoking about forty cigarettes per day for more than forty years. On the morning of the day before admission, he found that he was not able to read a book as his vision became suddenly distorted. He had additionally noticed weakness in his left leg during the daytime but went to bed without going to the hospital. The next morning, his weakness grew worse and he visited our hospital in a wheelchair due to gait disturbance. At this first admission, his NIH Stroke Scale (NIHSS) score was 4 points: hemiparesis 2, sensory disturbance 1, and dysarthria 1. He complained of diplopia when he looked to the right, but we could not observe his ocular deviation. His head MRI-DWI showed a scattered fresh infarction including the right occipital lobe, cerebral crus, and thalamus, and MRA showed occlusion of the P2 area of the right PCA and left SCA (). Carotid ultrasound (CUS) showed findings of moderate arteriosclerosis, and plaque accumulation in the carotid bifurcation was not observed. He was admitted to our hospital. Electrocardiogram (ECG) and monitoring data showed no findings of arrhythmia. We speculated that the cause of this stroke might be atherosclerotic change at first. We decided to begin injection of argatroban hydrate and edaravone, and administered oral aspirin 100 mg.\nEarly the next morning, a drop in consciousness level and aggravation of left hemiparesis presented. Emergency MRI was performed and showed enlargement of the lesion in the thalamus and additional cuneate fresh stroke on the right paramedian portion of the midbrain and small spotty fresh stroke on the left paramedian portion. MRA showed the extension of the occlusion of the PCA to the P1 portion (). At this point, we thought perforators branching from proximal part of PCA were occluded due to the progression of thrombosis in the PCA. We added cilostazol to avoid extra thrombosis. After half a day, his consciousness level began to improve, and we performed a detailed neurological examination. At this time, we observed bilateral ptosis. He could not open his eyelids on his own and showed compensated contraction of the frontal muscle. His ocular position was median; right eyeball movement was totally impaired and left could only perform slight adduction with ocular nystagmus. The right and left pupil diameters were 6.0 mm and 5.0 mm. Light reflex was not observed on the right and was observed only slightly on the left.\nDuring the course of treatment, transesophageal echocardiography (TEE) was performed to detect the cause of the stroke. It showed a patent foramen ovale (PFO) and noncompaction of the ventricular myocardium, which could have caused the stroke. According to the findings on TEE, we were convinced that the cause of the stroke was cardioembolic and changed his prescription from aspirin to rivaroxaban 20 mg.\nHis consciousness level improved gradually, and oculomotor paresis including ptosis only on the left side was also improved imperfectly 2 weeks later. Finally, disturbance of upward and downward abduction remained on the left side and severe oculomotor palsy also remained in the right side (). He could not achieve ocular convergence and Bell phenomenon in both eyes.\nIn addition, with regard to the motor paresis, we observed complete hemiplegia in the left upper extremity in the flexed position and mild hemiparesis in the left lower extremity. With the improvement of his consciousness level, he started standing and walking training with physical therapists. Although his muscle strength on the left lower extremity was sufficient, he had difficulty with those types of training due to ataxia of bilateral body trunk and lower extremities and attentional deficit as a higher brain dysfunction. Ultimately, he acquired walking ability with a cane, but he risked losing his balance and falling down when he had a lapse of concentration. With regard to higher brain dysfunction, mild cognitive dysfunction and memory deficit were present. At 12 weeks after initial presentation, he could leave our hospital and go home in relatively good condition. |
In 2015, a 22-year-old Chinese woman suffered a sudden loss of vision in her right eye with eye movement pain. She was diagnosed with “ON” and recovered after high-dose corticosteroid therapy in a local hospital. In April 2016, her visual acuity of the left eye suddenly declined to 20/100, accompanied by pain when the eyeball rotated. The same therapy was administered, but visual acuity was only slightly improved. Furthermore, there was recurrence in the right eye once, and there was recurrence in the left eye twice in the following 2 years. Unfortunately, the effect of corticosteroids worsened and the visual acuity became very poor.\nIn October 2018, when her left eye suffered again with visual acuity dropping to no light perception, corticosteroids did not work anymore. Finally, when there was recurrence in the right eye at the end of November 2018, visual acuity of both eyes dropped to no light perception. She then came to our ophthalmology clinic, with both eye blindness and pupil dilation; only her right eye had a mild light response. Fundus examination revealed pale binocular disc and miniature blood vessels. The patient had neither spastic weakness nor sensory signs of the legs and limbs. Visual evoked potentials examination indicated that the P100 waveform of both eyes nearly disappeared, while the retinal nerve fiber layer around the optic disc was significantly thinner than that of the normal in the optic coherence tomography assay (Fig. ). Magnetic resonance imaging (MRI, with contrast) showed no longitudinally extensive myelitis signals at the thoracic or lumbar spine, and there were no demyelination-enhancing changes in the brain, but some suspicious signals existed at the top right of the frontal lobe, which was considered ischemic lesions. Laboratory examination showed that the anti-aquaporin 4-antibody was positive in the cerebrospinal fluid (CSF, cell transfection).\nThe patient fulfilled the 2015 international consensus diagnostic criteria for NMOSD.[ She was treated with intravenous methylprednisolone 0.5 g/day for 5 days, then reduced to 0.25 g/day for 3 days, followed by daily reduced prednisolone start with 50 mg oral administration.\nFurther examinations showed that antibodies associated with SS were positive [anti-nuclear antibody (ANA) (titer, 1:1000), anti-SSA antibody, anti-SSB antibody, anti-Ro-52 antibody]. Complement C3: 0.77 g/L. Labial gland biopsy showed partial acinar atrophy of the salivary gland tissue and focal aggregation of interstitial lymphocytes (2 foci, > 50 lymphocytes per foci) (Fig. ). The rheumatoid factor (RF), erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP) were negative. However, there were no common symptoms of SS, such as dry mouth and eyes. Dry eye examinations were performed as follows: Schirmer - I (Sch- I) test results showed 4 mm for both eyes, tear breakup time (TBUT) was 3 s for the right eye and 6 s for the left eye, and the tear meniscus height was 0.11 mm for the right eye and 0.15 mm for the left eye. Although the patient did not have any obvious dry eye symptoms, she fulfilled the 2016 American College of Rheumatology (ACR)/The European League Against Rheumatism (EULAR) Classification Criteria of SS. Hence, she was diagnosed as “1. NMOSD; 2. SS.” On the basis of corticosteroid treatment, she was administered cyclophosphamide (CTX 0.4 g/day) intravenous drip once per week for 2 weeks and intravenous immunoglobulin (17.5 mg/kg/day) daily for 5 days. Her visual acuity returned to the binocular hand move after 5 days of treatment. Four weeks later, the patient’ s visual acuity increased to 20/800 in the right eye and finger counting in the left eye. |
An 8-year-old boy was brought with chief complaints of weakness over the lower limbs progressing to the upper limbs for 4 days and difficulty in breathing for 2 days. On admission, the patient's pulse and blood pressure were normal, but the patient had difficulty in breathing due to involvement of respiratory muscles. On central nervous system examination, the patient was conscious and oriented, there was hypotonia in all four limbs, the power was 2/5 on both shoulder muscles and was 0/5 in all other muscles acting on other joints, deep tendon reflexes were absent, and plantar reflex was not elicitable.\nThe patient had symmetrical ascending paralysis involving respiratory muscle, so clinical diagnosis of GBS was made and was corroborated by nerve conduction velocity test which was suggestive of sensory-motor polyneuropathy.\nThe patient was put on a ventilator and given IVIg 2 g/kg over 5 days. In view of no improvement, a repeat dose of IVIg 2 g/kg over 5 days was given. The patient required prolonged ventilation; therefore, tracheostomy was done. After 14 days of the second course of IVIg, no improvement was observed, so seven cycles of plasmapheresis were done. Following which, the child showed improvement in the power of respiratory muscle, and slowly, the patient was weaned off from a ventilator. There was an improvement in power of the upper limb muscle, followed by lower limb muscle. Gradually, the patient continued to improve; hence, the patient was weaned off from oxygen, and tracheostomy tube was removed.\nAfter 1½ months of admission and almost 1 month after IVIg therapy and 15 days after the last plasmapheresis cycle, the child had five episodes of convulsions generalized in nature. Following which, the patient had altered sensorium, inappropriate talks, and complete loss of vision. Blood pressure was normal. Clinical diagnosis of acute disseminated encephalomyelitis and PRES was made. MRI brain showed a bilateral asymmetrical area of abnormal enhancement in the parieto-occipital lobe with patchy areas of restricted diffusion at places suggestive of PRES []. The patient was started on anticonvulsants, following which, on the 2nd day, improvement in sensorium and vision was noted. Physiotherapy was continued, and as there was no new episode of convulsions, anticonvulsant was tapered off. Gradually, there was an improvement in power over a period of time; hence, the patient was discharged. After 3 months, MRI brain was repeated which was suggestive of normal study []. |
A 42-year-old male patient came to the Department of Conservative and Endodontics with a complaint of fractured amalgam restoration in lower left back region of the jaw.\nPatient was apparently alright 1 month back. Suddenly, he noticed fractured amalgam restoration in lower left back region of jaw. The patient had also given the history of burning sensation on the left and right cheek for the past 1 month. A detailed history was taken.\nOral examination revealed the presence of a white lesion on left and right buccal mucosa, adjacent to the amalgam fillings []. The lesions measured approximately 12.5 mm in length at the level of occlusal plane of the occluding molars extending from the region corresponding to first premolar to the second molar on the left side and 11.5mm on the right side. The lesion showed a reticular pattern with a reddish inflamed area surrounding it. The lesions were nonscrapable and tested Candida negative.\nThe patient had silver amalgam restorations on teeth no. 36, 37, 38, 47.\nThe patient's oral hygiene was fairly good. A cutaneous patch test was done to detect contact hypersensitivity. Alloy powder and mix were tested separately on skin on the back of the patient. The patient reported back after 48 h with the complaint of itching on the mix patch [Alloy + Hg]. Patches were removed and examined. A slight erythematous reaction was noted on mix patch area.[]\nTo confirm the diagnosis biopsy of the lesion and immunofluorescence assay was done.[]\nA clinical diagnosis of the OLR was made.\nThe patient was informed of the condition. It was decided to replace amalgam restoration with a nonmetallic interim restoration and follow up was advised.[]\nThe replacement of amalgam restorations with interim restorative material in left lower arch is done using Type II glass ionomer cement. The patient was asked to report after 1 month. On clinical examination, there was a reduction in size and severity of the lesion only on left side. But lesion on the right side was sustained []. Hence, the replacement of amalgam restoration in lower right arch was also done using Type II glass ionomer cement. One more review conducted after 3 months revealed complete healing of lesion on left buccal mucosa and reduction in size and severity of the lesion on right buccal mucosa was observed. Follow-up for 1 year was taken and complete healing of the lesions on both right and left buccal mucosa was observed []. |
A 44-year-old Chinese man, weight 55 kg and height 165 cm, presented to the neurosurgery department of our institution with an 11-month history of paroxysmal headache and weakness of the right limb without any other pertinent positive symptom. The patient had been diagnosed with NF1 for more than 30 years. His past surgical history included having undergone left parietal tumor resection in May 2016, the postoperative pathology of which indicated glial sarcoma (World Health Organization grade IV). In July 2017, the patient presented with paroxysmal headache with weakness of the right limb. Computed tomography (CT) of the head revealed recurrence of left parietal glioma, acute cerebral infarction in the left frontal lobe and around ventricle. As a result, the patient received conservative treatment in the neurology department.\nThe only pertinent positive physical examination finding was multiple cutaneous neurofibromas and numerous café-au-lait spots of different sizes dispersed over the trunk and limbs (Fig. ). The results of the patient’s cardiovascular, respiratory, and abdominal examinations were all unremarkable. His baseline blood pressure was in the range of 120–140 mmHg for systolic blood pressure and 65–85 mmHg for diastolic blood pressure. He denied any current medication, tobacco, or alcohol use, and a family history of NF was also excluded.\nThe results of the patient’s preoperative laboratory examination were unremarkable, including blood and urine analysis. His electrocardiographic examination showed normal sinus rhythm. His chest x-ray revealed multiple nodules in the two lung fields, the largest being located in the upper right quadrant of the lung field and having a diameter of about 29 mm. Magnetic resonance imaging of the head revealed the recurrence of glioma. The patient was scheduled for resection of the recurrent gliomas.\nDuring the preoperative examination, it was thought that general anesthesia application would be more appropriate for the patient. The patient was monitored with electrocardiography, heart rate (HR), invasive blood pressure, and pulse oximetry in the operating room. He was anesthetized with midazolam 3 mg, etomidate 14 mg, cisatracurium 20 mg, fentanyl 0.15 mg, and propofol 60 mg in sequence. When assisted respiration was started, the monitor showed a persistent increase in blood pressure. Within 40 seconds, it rose to about 310/140 mmHg, and the HR increased to about 140 beats per minute (bpm). We quickly eliminated the following possibilities: taking the wrong medicine, blood pressure monitoring equipment malfunction, or problem with venous access. To prevent the cardiovascular complications, we took measures to control the patient’s blood pressure and HR with phentolamine 2 mg, esmolol 30 mg, and remifentanil 80 μg when the blood pressure was about 310/140 mmHg and HR was about 140 bpm. The blood pressure values were stable during intubation, but the HR continued to be higher than 110 bpm.\nThe patient’s blood pressure was stable during the operation. However, there was a fluctuation of blood pressure during extubation with an increase to as high as 210/140 mmHg, which was aborted with phentolamine 1 mg. Nevertheless, the patient’s HR continued to be higher than 120 bpm, and he was not sensitive to β-adrenergic blockade.\nDuring postoperative follow-up, no headache, nausea, or blood pressure change (especially hypotension) was observed, and the patient’s tachycardia disappeared 3 days after the operation. To investigate the causes of severe hypertension during anesthesia induction, we initiated a biochemical workup of his adrenal hormone 3 days after the operation, which revealed elevated 24-hour blood laboratory test results: epinephrine 3.57 nmol/L (normal range, 0.01-0.34 nmol/L), metanephrine 8.99 nmol/L (0.01–0.42), normetanephrine 2.25 nmol/L (0.01–0.71), and vanillylmandelate 160.41 nmol/L (0.01–62). Subsequently, CT of the abdomen with contrast enhancement (Fig. ) demonstrated a 7.7-cm × 6.7-cm heterogeneous mass in the left adrenal gland. |
A 69-year-old male was admitted to a hospital in June 2016 because of right arm asthenia and dysarthria and was diagnosed as having cerebral infarction in the left middle cerebral artery area along with deep vein thrombosis. ECG demonstrated normal sinus rhythm and echocardiography revealed no intra-cardiac thrombus or vegetation. The patient was discharged from the hospital following administration of apixaban.\nIn August 2016, the patient was readmitted to the hospital because of recurrent right arm asthenia and dysarthria. MRI revealed multiple cerebral infarctions in not only the bilateral cerebral hemispheres but also the cerebellum. Trousseau’s syndrome was suspected at this time. Apixaban administration was stopped and an intravenous drip of heparin was started. Echocardiography revealed mild mitral regurgitation with vegetation on the mitral valve. Although the laboratory data suggested no evidence of infection, ceftriaxone and gentamicin were added as a precaution against infective endocarditis. The patient was then referred to our hospital for surgery.\nA CT scan revealed a left renal infarction and multiple swollen lymph nodes around both the abdominal aorta and stomach with antral hypertrophy, suggesting an advanced gastric cancer or lymphoma. As the vegetation showed no change despite the heparin and antibiotics therapy, cardiac surgery was performed on day 5 after referral. Extracorporeal circulation was instituted employing aortic and bicaval cannulation. After aortic cross-clamping, the mitral valve was exposed via a left atriotomy. Both mitral leaflets had vegetation on the surface, and major vegetation 15 mm in width was evident on the anterior leaflet (Fig. ). These were resected in their entirety and replaced with a 25-mm Epic bioprosthesis (Abbott). Continuous intravenous heparin administration was resumed on the following day, aiming for an activated partial thromboplastin time of between 40 and 50 s. Histologic analysis revealed that the vegetations were thrombi covered with vascular endothelium and that the mitral leaflet tissue was not damaged (Fig. ). On the basis of these findings, the patient was diagnosed as having NBTE.\nAn endoscopic stomach biopsy was performed on the seventh postoperative day, and histologic analysis revealed non-solid poorly differentiated adenocarcinoma with components of signet-ring cell carcinoma and moderately differentiated tubular adenocarcinoma. The patient was definitively diagnosed as having Trousseau’s syndrome and, subsequently, transferred to the department of surgery. A Billroth I distal gastrectomy was performed, and a continuous intravenous heparin drip was employed during the operation. Histologic analysis revealed poorly differentiated adenocarcinoma with a component of moderately differentiated tubular adenocarcinoma and metastatic tumor cells in the dissected lymph nodes (T4aN3bM0; stage IIIb). Further histologic analysis using alcian blue staining confirmed the presence of mucin in the tumor.\nSubcutaneous heparin injection was introduced on day 8 after the gastric surgery, and the patient was discharged from our hospital in October after acquisition of the self-injection technique. During this long hospitalization, no thromboembolic events were observed. Chemotherapy was started in November. The patient has survived for 18 months after the diagnosis of Trousseau’s syndrome without any recurrence of thromboembolism. |
Our patient is a 31 year old male teacher, who was brought in by ambulance to the Emergency Department. Two weeks earlier he had sustained an injury to the right side of his neck due to a rugby tackle, with a fierce "hand off" to the jaw extending his neck and rotating it to the left. He continued to play the game without any ill effects. A week prior to admission he was seen by his family physician and given an antibiotic for "swollen gland" in the right side of his neck. On the day of presentation he had sudden onset of right sided neck swelling with pain in the neck and shortness of breath.\nHis vitals were stable (Pulse 54, Respiratory Rate 24 and O2 saturation of 100%). He was conscious with GCS of 15/15. There was an obvious right sided neck swelling with trachea deviated to the left without any stridor. There was no neurological deficit or any evidence of Horner's syndrome. Our initial clinical impression was that he had a ruptured aneurysm.\nHe was promptly intubated in resuscitation rooms and taken for CT scanning. CT angiogram of the neck vessels showed active extravasation of contrast from his right CCA (Fig. ) resulting in the formation of a large haematoma extending superiorly into the parapharyngeal space and inferiorly into the superior mediastinum. It resulted in compression and marked displacement of the airway to the left (Fig. ). The thyroid gland was also displaced and Internal Jugular Vein was effaced due to compression. Our impression was that he had ruptured pseudo aneurysm of the Right CCA and he was brought forward for emergency surgery.\nHis neck was opened on the right side using a lazy S incision. On opening his platysma, there was a bulging haematoma which was rapidly expanding. Control of the Right common carotid was obtained proximally at the level of sternal head of sternomastoid by deflecting it, and distally at the level of bifurcation of common carotid. Omohyoid muscle was also divided for access. He was heparinised and the right CCA was clamped above and below the site of injury. Haematoma site was exposed by reflecting the internal jugular vein laterally and taking its multiple branches medially between clips. Vagus nerve was identified and protected. The point of perforation was in the posteromedial aspect of the artery, with surrounding adventitia appearing somewhat ragged, suggesting a stretch injury (Fig. ). This appearance was consistent with rupture of a pseudo aneurysm. The arterial perforation site was extended in a cranio-caudal fashion to expose normal vessel wall. Examination of rest of the arterial wall appeared relatively normal with no evidence of true or mycotic aneurysm. Primary closure of the extended defect was carried out with polypropylene 6-0 suture in two layers (first layer – full thickness wall, second – periadventitia as reinforcement). The sternal head of sternocleidomastoid muscle was reattached. |
The patient involved provided written consent for reporting of this case.
A 63-year-old woman with medical history of super morbid obesity (BMI 54) and atrial fibrillation for which she was anticoagulated with apixaban presented for an open reduction internal fixation (ORIF) of an ankle fracture. Significant medical history included diabetes mellitus type 2, obstructive sleep apnea, chronic obstructive pulmonary disease, and diastolic heart failure. The patient's last dose of apixaban was 48 hours prior to surgery. Other than moderate anemia (hemoglobin 8.8 g/dL), all laboratory studies, including a coagulation profile, were normal.
Prior to surgery, the patient was offered a sciatic nerve catheter and an adductor canal block as part of a multimodal postoperative analgesia strategy. Because of her many, serious medical conditions, we concluded that a peripheral nerve block offered the best opportunity to provide satisfactory postoperative analgesia. Specifically, we were concerned that the postoperative pain management primarily with opioid medications would pose increased cardiopulmonary risk to the patient. We were careful to explain the risks associated with peripheral nerve blocks, including the risk of bleeding and hematoma formation, and verbal consent was obtained.
The surgery was performed under general anesthesia and her intraoperative course was uncomplicated. Upon arrival to the recovery room, our acute pain service was contacted to evaluate her for peripheral nerve blockade. We positioned the patient in the lateral decubitus position and placed a sciatic nerve catheter. Though technically challenging due to body habitus, this sciatic nerve block was performed successfully and without any complication. The patient was then positioned supine for the adductor canal block. The leg was externally rotated and the knee slightly flexed for optimal positioning. A high-frequency linear array ultrasound transducer was applied to the mid-thigh in short-axis and the adductor canal was identified. Imaging was again challenging given the patient's habitus, but with firm compression of the ultrasound transducer, the important anatomical structures were clearly identified. The superficial femoral artery (SFA) was visualized |
A 18-year-old male patient was referred to our outpatient clinic because of masslike lesion in his left calf. He had a history of car accident and left isolated and closed tibia fracture 30 months ago that was treated by open reduction and internal fixation with 4.5mm broad DCP plate (, ). The surgical approach was through 1 cm lateral to tibial crest and plate was in lateral aspect of tibia. In this period, the patient was fully asymptomatic and had no complaint in routine follow-up. The patient had no significant past medical history. After 2 years, union was obvious in AP and lateral radiographs, and removal of tibial plate was decided. The plate had been removed 7 months ago on patient’s request. At the time of surgery, we used the previous incision and found that the bone was completely united. Plate and screw removal was done without any complication (eg, plate or screw break down).\nAfter the operation, the bone in radiograph was completely united and the patient was discharged when we find near 50-100 cc collection in the drain at the second day post operation.(). Examination at this time showed he was intact in neurovascular examination.\nAt the first post operation visit, he was asymptomatic and progressive partial weight bearing had been described. About 4 weeks after plate removal, he came back with mild paresthesia in the left foot at the plantar surface. Also, he had firm, pulse less and non-tender swelling with unclear border on the posterior aspect of the leg which gradually increased over time. Position of ankle was plantigrade.\nIn vascular examination, we found the dorsalis pedis and the posterior tibialis pulses were equal to the opposite leg. We followed this patient without further investigation.\nAt about 3 months after plate removal, paresthesia became persistant. We prescribe EMG/NCV for the patient. This assessment showed tibial nerve axonal injury pattern. Thus, we thought that paresthesia was due to iatrogenic tibial nerve injury (eg, bennet tip). Approximately 5 months after surgery, he returned with calf muscle firmness and occasional spasm like pain. Paresthesia remained mild and distal pulses were normal and swelling was more firm and fixed.\nIn physical exam, the skin over the mass was intact and we didn’t see ecchymosis or discoloration in this area. We found no lymphadenopathy. We found no tenderness at the fracture site and patient didn’t compliant from pain similar the previous fracture. Neurologic examination revealed normal force but decreased sensation in this mentioned area. We heard bruit over the surface of this mass.\nMRI was requested for this patient for calf muscle evaluation. At 1 month after MRI request, the patient came back and discovered a large (5 x 7 cm) soft tissue mass that was enhanced with probable diagnosis of hemotoma or pseudoaneurysm (-). Ultrasonography showed the pseudoaneurysm and an angiography was planned. After the patient was systemically heparinized with 5000 units of unfractionated heparin, angiography showed the widening between the anterior and posterior tibial artery which confirmed the pseudoaneurysm of the posterior tibial artery (, ). Thus, we proceeded for a common surgery with a vascular surgeon.\nDuring the common surgery, clots and hemotoma was evacuated and the posterior tibial artery perforation was repaired (, ). |
A 79-year-old female was admitted after a first seizure. She had a past medical history 12 years prior of an unruptured left MCA aneurysm treated with endovascular treatment with bare platinum coils. The patient had no past medical history of HBP, DM, vasculopathy or coagulopathy, and no history of smoking. No family history of ruptured/unruptured aneurysm was reported. MR and magnetic resonance angiography images before treatment showed a 10 mm left MCA aneurysm with minimal surrounding PE (, ). Sagittal T1-weighted images suggested the presence of a hyperintense intraluminal clot within the aneurysm (). On neuroimaging performed 2 days after endovascular embolization with coils, no signs of residual aneurysmal sac perfusion were observed on 3D-time of flight (TOF) images (), and the amount of PE was stable. The coiled aneurysm and the surrounding vasogenic edema remained unchanged over several imaging follow-ups. However, MRI performed 4 years after the initial treatment showed increased PE without evidence of aneurysm recanalization (, ). On a follow-up MRI, 8 years after the initial treatment, the aneurysm demonstrated a slight enlargement in size. The surrounding vasogenic edema was also mildly increased from prior studies, despite the absence of intraluminal flow on angiographic 3D-TOF images (, ). In addition, a new focus on the T1 hyperintense signal was observed along the aneurysm fundus on both TOF and T1-weighted images, reflecting new intraluminal clot formation (–). Because of the absence of aneurysm recanalization and the evidence of new clot formation, we interpreted the subtle enlargement of the aneurysm as a result of aneurysm-wall remodeling. The coil mass within the aneurysm lumen did not show any significant change in morphology compared to previous studies.\nMRI on admission, almost 12 years after endovascular treatment, showed markedly increased vasogenic edema extending to the white matter of the left temporal and parietal lobes, insula, and internal capsule with mild mass effect on the temporal horn of the left ventricle. The left temporal gyri appeared swollen.\nPerianeurysmal vasogenic edema has been rarely reported in association with small aneurysms in association with vessel-wall enhancement [,]; therefore, we decided to complete the MRI study with “black-blood” T1-SPACE (Sampling Perfection with Application optimized Contrasts using different flip angle Evolution) pre- and post-contrast sequences with isotropic 2×2×2 mm3 size. Vivid intraluminal and aneurysmal wall enhancement was observed on the post-contrast T1-SPACE sequence, with no signs of aneurysm recanalization on 3D-TOF images, in keeping with the presence of ongoing inflammatory phenomena within the coils-clot complex (–). No additional treatment was prescribed at this time, as the patient was already treated with corticosteroids for a long-standing medical history of polymyalgia rheumatica. A follow-up MRI study performed after 1 year from the onset of seizures demonstrated stable intraluminal and peripheral aneurysm wall enhancement and unchanged PE, suggesting persistent active inflammation of the intraluminal thrombus. |
A 50-year-old man with a history of liver dysfunction was admitted to Iizuka Hospital with the chief complaint of a prolonged fever and general fatigue in October, 2005. The temperature was over 38°C. Hematological laboratory data on admission were as follows: WBC 9670/ μl, ALP 552 U/l, CRP 16.7 mg/dl, and AFP 11.1 ng/mL. HBs-antigen was positive (). A Computed tomography (CT) showed a peripherally enhanced low density mass 7.5 cm in diameter, which located in segment 6 in the right lobe of the liver (). The tumor was accompanied with tumor thrombus to the posterior branch of portal vein (). Celiac angiography showed this lesion was hypervascular () and portal vein tumor thrombus in posterior segment was observed in the portal phase ().\nBiopsy was not done in this case because of danger of tumor seeding. Patients have chronic hepatitis B and then tumor was hypervascular and accompanied with portal thrombus, which is the feature of HCC, therefore a preoperative diagnosis was made as HCC with portal vein thrombus and nonsteroidal anti-inflammatory drugs were used to reduce the fever. Enough explanation was performed to the patient and informed consent for operation was obtained. Hepatic resection was conducted in November, 2005. The tumor was white and intrahepatic metastasis was observed (). A single nodule of disseminated cancer cells was found near the posterior segment on the abdominal wall during the operation. Posterior segmentectomy of the liver and thrombectomy including the dissemination was performed. On the cut section, the tumor was white and contained coagulation and necrosis (). Histological findings revealed poorly differentiated HCC with a trabecular pattern. Multiple tumors of intrahepatic metastases and portal vein invasion were observed. Neutrophil infiltration in the tumor was observed (). The noncancerous liver tissue showed a moderate chronic inflammatory infiltrate in the fibrous stroma, diagnosed as liver fibrosis. Immunohistochemical stain of IL-8 was performed with paraffin embedded sections of liver tissue using mouse monoclonal antibody at dilutions of 1 : 100 (Assay designs, Ann Arbor, USA). Immunohistochemical staining was described previously []. The subsequent reaction was performed by the peroxidase labeled streptavidin-biotin technique using Histofine SAB-PO kit (Nichirei, Tokyo, Japan). IL-8 was observed in the cytoplasm of cancer cells ().\nAfter the tumor resection, fever disappeared and CRP dropped gradually from 16.7 to 2.7, but the patient had recurrence with multiple liver metastasis and pleuritis carcinomatosa and died on February 2, 2006, 79 days after the operation. |
An 88-year-old male presented with a recent history of acute abdominal pain and vomiting. His past medical history included ischaemic heart disease, cerebrovascular accident and an open repair of abdominal aortic aneurysm 10 years earlier, in addition to an unoperated incisional hernia. On examination, the patient was septic and in cardiogenic shock. Urgent CT abdomen and pelvis showed free intra-abdominal air (Figure ), thickened descending and proximal sigmoid colon with locules of transmural air, as well as locules of extra-luminal gas around it, and this was thought to be the site of the perforation. Because of the presence of aortobiiliac stent within the abdominal aortic aneurysm, origin of the inferior mesenteric artery could not be delineated. However normally opacified tributaries of inferior mesenteric artery supplying the sigmoid and descending colon via collaterals were demonstrated (Figures , , ). The patient had undergone an emergency laparotomy, adhesiolysis and subtotal colectomy with an end ileostomy. Intra-operative findings included friable large bowel from mid-sigmoid to caecum with a widespread ischaemic appearance and multiple perforations. Postoperatively, the patient required intensive care unit admission, and unfortunately, he died a week later due to sepsis and multiple organ failure.\nPathological examination of the resected bowel revealed colectomy with terminal ileum specimen; the appendix was not identified. On opening the colon, there were extensive but patchy areas of mucosal ulceration with elevated edges. The margins of the ulcers appeared elevated. A focus of colonic perforation was identified. No tumours were identified macroscopically. The mucosa was extensively sampled sequentially from the ileum through the distal colon to include both resection margins. Microscopic examination of the ileum appeared normal, while sections from the large bowel showed extensive areas of mucosal ulceration with features of transmural acute ischaemic necrosis and associated suppurative serositis. Arteriosclerotic vascular changes in several blood vessels with some vessels showing marked luminal narrowing supportive of ischaemic colitis are noted. A single 8mm polyp of mainly poorly differentiated adenocarcinoma in addition to well-differentiated adenocarcinoma that had invaded into the pericolic adipose tissue (pT3) was found in a single section from the ascending colon. The poorly differentiated adenocarcinoma was present in both the mucosal surface and deeper layers of the colonic wall beyond the muscularis propria (Figure , ). The full extent of tumour invasion by the poorly differentiated component was highlighted by pancytokeratin (MNF116) immunostaining (Figure , ). CDX2 was positive in the well-differentiated component of the tumour but uniformly negative in the poorly differentiated aspect (Figure ). In the pericolic fat, the tumour cells were differentiated from macrophages with CD68 that stained the macrophages (Figure ). The poorly differentiated component of the tumour in our case showed negativity for CK7 uniformly. CK20 showed patchy positivity in the superficial well-differentiated component but was uniformly negative in the poorly differentiated component. The primary colonic origin of the adenocarcinoma, especially the poorly differentiated component, necessitated immunostaining with CK7 (Figure ), which was negative. CK20, which is an epithelial marker with restricted expression compared to CK7, revealed very occasional positive tumour cells in the well-differentiated component (Figure ), but was uniformly negative in the poorly differentiated component (Figure ). Special AT-rich sequence-binding protein 2 (SATB2), a useful new highly specific biomarker, is expressed in 85% of all colorectal carcinomas. In combination with CK20, it identifies more than 95% of all colorectal cancers; it is recommended in lower GI tumours due to its high specificity. The above immunohistochemical pattern is consistent with the established practice from molecular studies that colorectal cancer is a heterogeneous group of neoplastic diseases that develop through three main pathogenetic pathways, namely the chromosomal instability pathway, the microsatellite instability (MSI) pathway, and the CpG island methylation pathway. Clinicopathologic studies are also pointing to colorectal carcinomas originating through these three pathways to differ in the type of precursor lesions, natural history, and pathological features. The combination of CK20/CK7 immunoprofiles showed that the CK20+/CK7− profile was the highest (60.4%), CK20−/CK7− was 35.4%, CK20+/CK7+ was 2.1% and CK20−/CK7+ was 2.1%. CK20 is specific for colonic, urothelial and Merkel cell carcinoma. On the other hand, CK7 is characteristic of glandular malignancies originating from the breast, respiratory tract, biliary tract and Mullerian epithelium [].\nSo CK7−/CK20− pattern as seen in the tumour we report is a known phenotype for colorectal cancer, which is now being shown to be heterogeneous in its molecular profile and therefore patient outcome []. CDX2 immunostaining was diffusely positively in the well-differentiated tumour component, while it was negative in the poorly differentiated component (Figure , ). CDX2 is a homeobox gene comparatively specific for intestinal epithelial tissue [].\nThis tumour was shown by immunohistochemistry to be MMR protein deficient (MHL1 & PSM2 negative) (Figure , , , ), but MSH2 (Figure , ) and MSH6 (Figure , ) positive. The MLH1 promoter hypermethylation was detected in the tumour-derived DNA component of the sample and within the spectrum of MSI-H and/or mismatch repair deficient (dMMR by IHC) primary colorectal adenocarcinoma, and all these results indicate a somatic process. However, these results do not exclude Lynch syndrome in a patient with a strong family history (Amsterdam II Criteria), in which germline DNA testing may still be considered necessary. This case is regarded as a de-novo condition as there is no relevant previous family history; this is significant for the other related family member. |
We presented a 20-year-old male patient, who fell from a height of about 30 feet. He was initially managed at a nearby community hospital where he was diagnosed to have multiple rib fractures on both sides, with bilateral hemopneumothorax. The primary management of patient was performed with bilateral intercostal chest drains and positive pressure ventilation for lung contusion. The patient had no neurological deficit at initial presentation. He was then referred to our center after four weeks for further management.\nOn examination, there was tenderness over the tenth thoracic vertebrae with mild knuckle deformity. There was no motor or sensory deficit at any level. Superficial and deep tendon reflexes were normal. Radiographs and computerized tomography showed a fracture dislocation with spondyloptosis of the ninth thoracic vertebra (T9) over the tenth thoracic vertebra (T10) with vertebral body fracture of the eight vertebral body ( - ). The pedicles of both T9 and T10 vertebrae were fractured bilaterally, thus separating the posterior elements from their respective vertebral bodies. There was complete spondyloptosis of T9 over T10 vertebral body and both T9 and T10 vertebral bodies could be seen in a single transverse section of computerized tomography (). The patient was scheduled for surgery after improvement in general and lung condition. The spine was approached through standard posterior midline incision. There was no significant kyphosis seen and the posterior elements of the eighth and ninth thoracic vertebrae were lying almost in place with undisplaced fractures in the lamina of respective vertebra. The pedicle screws were inserted in the fifth, sixth and seventh thoracic vertebrae proximally and the tenth, eleventh and twelfth vertebrae distally ( - ). An in-situ posterior instrumentation with laminectomy of T8 and T9 vertebrae and posterolateral fusion from the fifth to twelfth thoracic vertebrae was performed. No attempt was made to reduce spondyloptosis of T9 over the T10 vertebrae. The patient tolerated the operation well and there was no postoperative neurological deterioration. He was mobilized with the help of customized dorsolumbar rigid orthosis on fifth postoperative day. He was followed up at monthly intervals and radiographs along with computerized tomography showed satisfactory in situ fusion between T9 and T10 vertebral bodies (). The patient returned to his previous occupation. |
A 40-year-old man was admitted to our hospital with pain in the right upper quadrant and epigastric region beginning 20 days before, after weightlifting and loss of appetite associated with nausea. The patient had no history of liver disease, jaundice, or changing in bowel habits. Fever appeared the last day before the admission probably because of partial rupture of the cyst as a result of abrupt increase in intraabdominal pressure and spillage of the content into the peritoneal cavity.\nAbdominal examination revealed right flank tenderness and abdominal enlargement with dullness on percussion without shifting dullness indicating either fluid collection or mass. Further diagnostic evaluation raised the clinical suspicion of abdominal malignancy. On the abdominal ultrasonography, there was a large >10 cm, solid, hyperechogenic, cystic lesion with heterogeneous pseudotumor appearance and presence of large amount of perihepatic, intraperitoneal, and pelvic ascitic fluid nonpathognomonic of hydatid disease. The gallbladder was normal, and no other lesions were seen. Results of laboratory tests except of a mild leucocytosis otherwise were normal. Abdominal computed tomography (CT scan) revealed the presence of a 25 cm × 21 cm × 14 cm cystic mass strongly adherent to the right hepatic lobe. The cyst filled the entire right and median side of the abdominal cavity, displacing the intestinal loops to the left, and was extended down into the pelvis. The patient was taken into operation with the diagnosis of a large cystic with solid features tumoral mass (). Upon direct examination, a huge thin-walled cyst, which grew exophytically from the anterior part of the right hepatic lobe containing daughter vesiculae, was found to occupy the entire right side of the abdomen. The pericyst was found strictly adherent to the parietal peritoneum and the bowel loops. Yellow, nonprulent gelatinous matrix with daughter cysts at various stages of degeneration was found in the cyst. The cultures that were obtained intraoperatively from this fluid were negative for bacterial infection. Microscopy of the cystic fluid showed hooklets of E. granulosus ().\nThe patient made an uneventful recovery and was discharged the 8th postoperative day on albendazole (400 mg twice daily) for two months. There was not recurrence of hydatid cyst at nine months follow up of the patient. |
An 82-year-old Caucasian woman presented to our clinic with a persistent vegetative state after a coma. She was admitted at the age of 73 years, one year after a stroke, to the neurologic rehabilitation department of a Bavarian hospital. The diagnoses at that time included motor hemiparesis, motor aphasia, an inability to speak, an inability to swallow and an inability to actively move or react to contact attempts. She groaned continuously and on neurological examination, both her pupils were round and reacted directly and indirectly to light. It was not possible to examine her coordination because of an inability to stand or walk. In addition, she had no reaction on sensibility tests. A sensory aphasia was assumed.\nElectroencephalography indicated a left-sided frontotemporal theta-delta state and no epilepsy-specific signs. Computed tomography indicated an extensive infarction and/or occlusion of the left middle cerebral artery and a widened left ventricle as well as a lowered density of her left basis pontis and brain stem. The reported laboratory values for her blood, liver and kidneys were in the normal range.\nTherapeutic modalities instigated at that time by that hospital included a continuation of anticoagulation therapy which had started one year prior. A urinary infection that was discovered was treated with norfloxacin (based on resistance tests). Any logopedic or physiotherapeutic action was considered to be fortuitous, as only passive physiotherapy was possible. A feeding tube was inserted because of the patient's inability to swallow. She was dismissed from that hospital after six weeks for care at home. It was assumed that she would survive only a few months and that she should live the last months of her life in the home environment. The nursing was to be administered by her relatives, all farmers, with a nurse coming every day. Unless a special event necessitated an earlier visit, a family doctor visited every two weeks.\nNine years after her initial stroke, the 82-year-old woman had passed into a deep sleeplike state that had already lasted seven months, as the woman became our patient. Patient contact was not possible, and there were few vocal signs, such as groaning or cries. Her limbs had to be passively moved, as no active physiotherapy could be induced. She did not change position in the bed by herself, and active movements with her arms or legs were not observed. The only remaining signs of patient activity were vocal signs such as uttering of the same word, 'Maria', or occasional groaning. She was continuously administered anticoagulant therapy and artificial feeding. A search for microbial infections indicated serologically significant immunoglobulin G and immunoglobulin A titers against Chlamydia trachomatis, Chlamydophila pneumoniae and a suspected reactivation of Epstein-Barr virus\nAfter isolation of immune cells from her peripheral blood, as described elsewhere [], the immune cells were incubated in vitro with the OKT3 anti-CD3 antibody (ORTHOCLONE, muromonab-CD3 human T cell blocker, Janssen-Cilag, Neuss, Germany), which activates T lymphocytes. The stimulated T lymphocytes activate other immune cells, mainly monocytes and dendritic cells, and produce neuroactive substances []. These in vitro-activated mononuclear immune cells were administered by intramuscular injection. Alternatively, the activated immune cells were used to activate other not yet activated immune cells in vitro. These 'cascade primed' immune cells better recognize and eliminate infected cells and cancer cells []. As described previously for the treatment of patients with schizophrenia or depression [], approximately 40 × 106 to 60 × 106 autologous activated immune cells were injected intramuscularly once per week.\nSeveral changes in the behavior of our patient could be observed after the initiation of ACT. After the first injections, our patient opened her eyes upon demand and moved them in the requested directions. Furthermore, our patient's eyes followed the person entering the room, and she was able to turn her head (Figure , month one). Other motor function improvements occurred more slowly, but our patient was able to change her side position in bed by herself and, after six weeks, she could grab for the bar of the bed with the hemiplegic-side hand (Figure , month two). Six months after beginning the immune cell therapy, a logopedist started training for the swallow reflex and soon claimed that our patient could swallow independently. The nurse had observed that our patient independently move her tongue during tooth brushing. Although our patient grabbed the hands of her grandchildren with both hands, held them for a lengthy period and looked at the children, this was not definite proof that she recognized them. Unfortunately, our patient died after a night-time tube feeding after aspirating vomit. |
A 71-year-old woman, gravida 6, para 4, with a two-week history of general malaise and no history of gynecological surgery, was referred to the department of cardiovascular medicine in our hospital since an echogenic mass in the right atrium was detected with cardiac ultrasonography. She had a history of hypertension, diabetes, and atrial fibrillation that had been well controlled by medication. Laboratory tests revealed normal results for tumor markers, liver and kidney function, and D-dimers, except for hemostatic abnormalities associated with anticoagulant drugs taken for atrial fibrillation.\nDynamic contrast-enhanced computed tomography (DCE-CT) of the chest and abdomen was performed to determine the extent of the tumor. Arterial phase contrast-enhanced CT (AC-CT) images indicated the presence of a hypodense uterine mass and dilation of the right uterine artery (RUA). Although it was difficult to trace the tumor arteries using only axial plane images, multiplanar reconstruction images along the tumor arteries enabled tracing of the tumor arteries through the route of the right ovarian vein. Several small arteries originating from the RUA were considered to be tumor arteries because they extended to the IVC through the right ovarian vein with increasing diameter and then extended into the right atrium and the right ventricle. A plexus of small arteries, which comprised complex vascular formations originating from the right ovarian artery, the subcostal artery, and the right uterine artery (), surrounded tumor arteries along the route of the right ovarian vein and caused the diameter of tumor arteries to expand by communicating with each other at many locations ().\nMagnetic resonance (MR) imaging was performed to assess the characteristics of the uterine tumor. On MR images, the uterine tumor, approximately 13 × 7 × 6 cm in size, extensively involved the myometrium as a poorly demarcated diffuse lesion. On T1-weighted images, the lesion exhibited low signal intensity (SI) similar to that of the myometrium and included a number of hyperintense tiny foci that were suppressed on fat-suppressed T1-weighted images, reflecting a lipomatous tumor. On T2-weighted images, the lesion exhibited inhomogeneous high SI and involved a few hypointense nodules. On contrast-enhanced fat-suppressed T1-weighted images, the lesion demonstrated strong contrast enhancement but contained several poorly demarcated hypointense areas, reflecting degenerative or necrotic changes. On diffusion-weighted images and the apparent diffusion coefficient (ADC) map, the lesion showed slightly high SI (). On the basis of these findings, lipoleiomyoma was considered to be the most likely diagnosis for the uterine tumor.\nThe presumptive diagnosis of intravenous leiomyoma (IVL) was made based on a combination of CT and MR findings, and single-stage surgery with deep hypothermic circulatory arrest was scheduled. The operation was performed with a team of gynecologists and cardiac surgeons. First, a skin incision was made from the pelvic symphysis to the xiphoid process via the left side of the navel, and the surgical wound was separated by a retractor (Lobster “Wishbone”-style retractor system). The uterine fundus was enlarged with smooth surface. Bilateral adnexa and omentum were inspected and found to be normal with the exception of mild omental adhesion to the anterior abdominal wall. The right infundibulopelvic (IP) ligament had thickened to 3 cm in diameter since plexiform arteries arising from the right uterine artery surrounded the ligament. Furthermore, the plexiform arteries formed a connection to IVC along the route of the right ovarian vein ().\nSecondly, a thoracotomy was performed, and a cardiopulmonary bypass was established with ascending aortic arterial return and venous drainage through the right atrial appendage. After the circulation was interrupted, systemic temperature was reduced to 22°C. The IVC was then opened below the renal veins. The intracardiac mass was found to be floating freely, with the exception of being slightly adhered to the internal surface of the right ventricle. While the cardiac surgeons separated the adhesion, the gynecologists incised the IVC via a venotomy and cut off the intracaval tumor at its origin. The intracardiac and intracaval components of the tumor were drawn out upward through the right atrial incision. The uterus was found to be enlarged with an intramural tumor. Moreover, a continuous plexus of small arteries was observed along the right ovarian vein from the uterine tumor to the IVC. Subsequently, transabdominal hysterectomy and bilateral salpingo-oophorectomy were performed, with the right ovarian vein surrounded by the plexus of small arteries removed as well. Through these procedures, complete tumor resection was achieved. She was discharged on the 18th postoperative day without any complications. Clinical and radiologic assessment over a 7-month follow-up has shown no evidence of tumor recurrence. Histopathologically, the uterine lesion was composed of variable amounts of smooth muscle and fat cells with hydropic and hyaline degeneration. An intravenous component was not identified in the resected specimen. The intravascular mass originating from the IVC and extending to the right ventricle was composed of degenerated smooth muscle cells with a number of large vessels. On immunohistochemical staining, the nuclei of smooth muscle cells in the tumor were positive for estrogen receptor (ER) and progesterone receptor (PR) (). |
We describe a case of a 54-year-old Caucasian female who presented with abdominal pain from her recurrent nephrolithiasis. She had a right 2.1 cm partial staghorn calculus and 1 cm left lower pole calculus. She underwent right PCNL. Under fluoroscopic guidance, the right upper calix was accessed through an intercostal approach between the 11th and 12th ribs and had a right percutaneous nephrostomy tube placed at the end of the procedure to maintain percutaneous renal access for possible second look nephroscopy. Ureteral stent was not placed as there was no indication for stent placement especially that nephrostomy tube was placed. There were no immediate postoperative complications, and a routine postoperative chest radiograph was unremarkable. The next day, the patient complained of dyspnea and worsening pain. Routine postoperative computed tomography (CT) scan showed two small residual stones in right ureter in addition to the previously known left renal stone. A chest X-ray done on postoperative day (POD) 2 was unremarkable except for atelectasis. The pain management service was consulted. For complete stone removal, she was taken to surgery on POD 3 for a second-look cystoscopy with bilateral ureteroscopy. Residual stones were removed from the right kidney along with the left renal stone. A left ureteral stent was placed at the end of the procedure. No stent was placed in the right ureter as she already had the nephrostomy tube in place, the right ureter was patent at the end of right ureteroscopy with minimal traumatization, there was no suspicion for any pleural injury at that time, and we did not want the patient to be bothered by having bilateral stents. The patient's pain improved and the patient was discharged from the hospital on POD 4 after removal of her right percutaneous nephrostomy tube. She was discharged with the indwelling left ureteral stent in place with removal planned in 1 week in clinic.\nThe patient was readmitted a day later for respiratory distress that required supplemental oxygen through a nonrebreather face mask. A CT scan of the chest, abdomen, and pelvis with contrast showed a large right pleural effusion with a fistulous tract between the right kidney and right pleura ( and ). The patient expressed serious reservations about pain control given her difficulties in the previous admission, and preferred less invasive options when management strategies, including tube thoracostomy, were discussed. Emergent therapeutic thoracentesis was performed instead and 2000 cc of pleural fluid was drained. The patient's respiratory distress significantly improved after thoracentesis. Pleural fluid analysis showed low fluid pH, and a fluid-to-serum creatinine ratio of around 4.5, both consistent with urinothorax.. The patient had another thoracentesis on POD 6 from her initial surgery and 700 cc pleural fluid was again drained. On the following day, the patient returned to the operating room and a 6F, 26 cm Double-J ureteral stent was placed in the right ureter along with 16F urethral catheter to prevent urine reflux with voiding. The patient thereafter demonstrated gradual improvement in her symptoms without reaccumulation of fluid in the pleural cavity and was subsequently discharged home in stable condition. Her Foley catheter was removed 2 weeks later. Retrograde pyelography at 1 month follow-up showed complete resolution of her nephropleural fistula and the stent was removed at that time (). At about 6 months from the said visit, the patient has not subsequently had respiratory complaints. |
A 50-year-old male with a chief complaint of pain during chewing and swallowing was referred to our department He mentioned a history of trauma to his right mandibular angle during an assault four months previously. The patient did not have any neurosurgical signs, loss of consciousness (LOC), dizziness or vomiting. The Glasgow coma scale (GCS) of the patient was 15 when he was referred to our department.\nThe main complaints of the patient were pain on the right side of the neck and lower jaw during chewing and swallowing. Although the patient experienced pain during mandibular movements and yawning, no limitation of jaw movements was observed. The nature of the pain was sharp and intermittent and lasted a few seconds every time the patient turned his head or upon chewing. The maximum mouth opening (MMO) was about 46 millimeters and there was no deviation in mandibular movements. On clinical examination, there was neither extra-oral swelling nor tenderness of temporomandibular joint (TMJ) or masticatory muscles. Intra-oral examination did not show any occlusal problems or premature contact. The patient did not mention any surgical history or tonsillectomy. He did not complain of symptoms mimicking glossopharyngeal neuralgia.\nA panoramic radiograph was obtained from the patient (). The radiographic view of the jaws showed elongated SP in the left side whereas the right SP was normal in size but displaced. The right displaced SP on panoramic view suggested fracture of this process without mandibular condyle fracture. Computed tomography (CT) was obtained for more evaluation and to detect other possible fractures. The CT views confirmed right SP fracture ().\nThe treatment plan for the patient was conservative treatment and follow-up. He was instructed to restrict neck movements. An anti-inflammatory (Naproxen 500 mg for every 8 hours for two weeks) and muscle relaxant (Tizanidine 2 mg twice a day for two weeks) drugs were prescribed to reduce the pain during the mastication. After four months of observation, the patient’s pain and discomfort resolved and he was able to masticate and swallow painlessly. |
A 37-year-old female patient reported to us expressing concern over the mobility of her teeth, the presence of a recurrent fistula, and overall displeasure with the height of her gingival smile line (). Upon clinical examination, it was determined that the maxillary incisors (i.e., teeth numbers 7, 8, 9, and 10) were compromised and that horizontal bone resorption had occurred as a result of aggressive periodontitis. The right central incisor was extruded and dislocated due to secondary occlusal trauma, and the left central incisor possessed a horizontal root fracture (Figures and ). Intraoral periapical radiographs revealed horizontal bone defects in all four of the maxillary incisors (). Severe resorption of the alveolar ridge in the premaxillary area, along with complete resorption of the buccal and palatal bones adjacent to the roots of maxillary incisors, was also observed ().\nAfter apprising the patient of her situation, we offered a number of viable treatment options; however, the patient has adamantly opposed many of them. Because the patient did not wish to use dentures and wanted to retain the ability to floss between prosthetic teeth, we were restricted from implementing a number of conventional treatment options. Despite being constrained by both the extent of patient's periodontitis and her aforementioned wishes, we nevertheless outlined a treatment plan that conformed to the patient's desires and would restore the aesthetics and functionality of the compromised teeth. In agreement with the patient, it was decided that the compromised incisors be extracted and replaced with four short locking-taper implants using a minimally invasive surgical technique which would not require flap raising or bone grafting procedures.\nThe patient was treated with a local anaesthetic prior to extraction (4% Articaine with 1 : 100,00 adrenaline, Ubistesin R; 3M ESPE). Care was taken to extract the teeth with minimum trauma so as not to damage the buccal or palatal bone plates. The compromised teeth were luxated and extracted while avoiding lateral movement. Following extraction of the affected teeth, the implant sites were prepared using a 2.0 mm diameter pilot drill and a 2.5 mm drill on an 18 : 1 hand piece at 1000 RPM with constant irrigation (2.0 mm Standard Pilot Drill, Bicon LLC, Boston, MA). Using a 400 : 1 hand piece at 50 RPM, the osteotomies were produced by the sequential use of 2.5, 3.0, and 3.5 mm reamers (Latch Reamers, Bicon LLC, Boston, MA), followed by hand reaming with a 4.0 mm reamer (diameter of implant). Bone obtained from the reamers was stored in a silicone dappen dish for later grafting. The osteotomies were generated to a final depth that would result in the implant shoulders lying 2.5 mm below the alveolar crest. Four endosseous root-form short implants (4.0 × 8.0 mm MAX 2.5 Implants, part #260-340-008, Bicon LLC, Boston, MA) were then inserted using the manufacturers inserter and further tapped in using a seating tip (). The implant placed in position 8 was inserted in more vestibular position in order to accommodate its size and prevent interference between prospective prosthetics (i.e., prosthetic on the lateral incisor). Bone harvested during reaming, along with tricalcium phosphate (SynthoGraft Pure Phase Beta-Tricalcium Phosphate, part #260-400-150 Bicon LLC, Boston, MA), was applied to the shoulder of the implant; healing plugs were used to avoid the deposition of bone graft particles inside the implant well. The healing plugs were then replaced by preformed shouldered parallel abutments (Universal Stealth-Shouldered Abutment, Bicon LLC, Boston, MA) upon which polycarbonate snap-on sleeves (Temporization Sleeves, Bicon LLC, Boston, MA) were adapted to receive an immediate temporary restoration (Figures , , and ).\nThe immediate temporary restoration consisted of a nonfunctional temporary bridge, which was seated and adjusted to clear centric and eccentric contacts and to support the papillae without encroachment (). The implementation of a “snap-on” system between the abutment and emergence sleeve precluded the use of cementation, which in turn abrogated the potential for soft tissue irritation. Postoperative care included 2 grams of daily oral antibiotics for 6 days (Augmentin, GlaxoSmithKline, Verona, Italy). Additionally, the patient was given detailed postoperative instructions about analgesic therapy and oral hygiene, along with a 0.12% chlorhexidine mouth rinse to be administered 3 times a day for 7 days (GUM PAROEX Chlorhexidine Gluconate Oral Rinse 0,12% CHX + 0,05% CPC, Sunstar Suisse S.A., Etoy, Switzerland). Three weeks after surgery, CT scans confirmed implant position and were absent for signs of vestibular bone dehiscence (); furthermore, the patient exhibited good wound healing (with the exception of slight gingival recession at tooth number 8—see Discussion) ().\nThree months following the procedure, the temporary bridge was then replaced by two temporary prostheses (supported by implants in positions #7 and #8 and in positions #9 and #10) to guide the regrowth of the gingival contours (Figures and ). Four months following the placement of the temporary prostheses, we performed two connective tissue grafts to thicken peri-implant soft tissues promoting a more natural and aesthetic emergence profile. Connective tissue for the first graft was harvested from the left palatal mucosa and grafted to the implants in positions #7-8. Then, after a two-month healing period, tissue was harvested from the right palatal mucosa and grafted to the implants in positions #9-10 (Figures and ). Both tissue grafts were harvested without epithelial layer using the trap door technique. Three months after the second tissue graft procedure, the patient had healed sufficiently enough to allow the placement of the final restorations ().\nLateral/anterior protrusion of the definitive restorations was achieved using canine guidance: in this way, mutually protected occlusion prevented contact between incisors during all mandibular eccentric movements, and incisors came into contact with their antagonists only during maximum intercuspation.\nTo orient the seating of the final abutment, a jig was fabricated and utilized to aid in correct positioning. A direct impression was then taken using a polyether impression material (Impregum Penta, 3M ESPE, St. Paul, MN). We then prepared a stone cast using type IV extra-hard dental stone, from which the definitive abutment could be individually modified. Finally, four zirconia crowns were fabricated and cemented on the abutments using extraoral cement (RelyX Unicem, 3M ESPE, St. Paul, MN). The abutment and crown were then tapped through the long axis of the post into the implant well using a 250 g mallet (). |
A 56-year-old woman presented to our department with a chief complaint of pain in her left TMJ in December 2014. Around November 2012, the patient had noticed pain in her left TMJ when widely opening her mouth, but the symptoms disappeared, and she did not address the matter. Around July 2014, the symptoms recurred, and she began experiencing trismus. She visited the dental and oral surgery department at a hospital for examination. Both computed tomography (CT) and MRI indicated synovial chondromatosis of the TMJ, and she was referred to our department for treatment.\nClinical examination found that she had a fairly healthy physique and no signs of nutritional deficiency. There was no visible facial deformation, and the maximum mouth opening was 30 mm. Movement of the left mandibular condyle was poor, and there was pain as well as joint sounds when widely opening the mouth but no occlusal abnormality ().\nPanoramic radiography showed that the left mandibular condyle was flattened, with a radiopaque image near the superior joint cavity (). The TMJ radiograph also showed deformation in the left mandibular fossa. With mouth opening, both mandibular condyles were able to move to near the lowest point of the articular eminence. CT showed that the left mandibular condyle was flattened, and small and granular nodules were observed in the joint capsule of the left TMJ, some of which were calcified ().\nT1-weighted MRI of the left TMJ showed deformity of the left mandibular condyle and anterior disc displacement without reduction, and T2-weighted MRI showed fine nodular areas of hypointensity in the superior joint cavity (Figures –). Bone scintigraphy showed accumulation at an area matching the left TMJ (). Based on these findings, the final diagnosis was of left synovial chondromatosis of the TMJ.\nApproximately 2 months after the initial examination, the masses were removed, and plastic surgery was performed on the mandibular condyle under general anesthesia. Incision lines were set according to the Al-Kayat–Bramley method []. The skin incisions were made, detaching up to the temporal fascia. An incision was made in the superficial temporal fascia, then in between the superficial and deep layers to the zygomatic arch, where the periosteum was incised to expose the joint capsule. The joint capsule was punctured, and approximately 2 mL of slightly viscous synovial fluid was collected. Incising the joint capsule revealed numerous white loose bodies around the mandibular condyle and in the superior joint cavity. The loose bodies were not adherent to the surrounding tissue and were carefully removed.\nAlthough the articular disc was preserved, the mandibular condyle was deformed. Therefore, plastic surgery of the mandibular condyle was performed using an ultrasonic cutting instrument (Piezosurgery®, Mectron Medical Technology, Carasco, Italy). A closed drain was placed in the wound, and the incision was sutured to complete the operation ().\nAt the time of writing, it has been more than 5 years since the surgery. There have been no signs of local recurrence or trismus. Image findings have also not indicated any recurrence (Figures and ).\nThe loose bodies removed were 2 to 5 mm in diameter, white, bone-like, hard, and numbered 386 in total ().\nHistopathologically, there was proliferation of cartilage tissue with chondrocytes forming round or irregular small masses surrounded by fibrous tissue, along with fibrosis and ossification. There were no signs of malignancy, which is consistent with synovial chondromatosis (). |
A 65-year-old woman presented with left-sided neck pain and swelling since 5 days prior to admission. She reported progressive dysphagia during this period after a history of grilled fish ingestion. There was no pertinent past medical and family history, and she denied any drug or substance abuse. Physical examination revealed low-grade fever (T: 38°C oral) and mild tachycardia (HR: 95 bpm). There was marked stiffness of the neck with an 8 × 8 cm erythematous bulging anterior to left sternocleidomastoid muscle at the level of the thyroid cartilage. A spiral neck CT with IV contrast was done. A linear foreign body with horizontal orientation was seen in the superior part of the pharynx along with a 40 × 20 mm collection around it (). There was also thrombosis with an air bubble in the left internal jugular vein, and the distal end of the fishbone was adjacent to the left internal jugular vein. Due to the high risk of endoscopic removal, we decided to proceed to surgery for better exposure and also effective drainage of infection. Via a classic incision anterior to the left sternocleidomastoid muscle, hypopharynx and cervical esophagus were explored and the carotid sheath was opened. A 4 cm sharp fishbone was found impacted transversely into hypopharynx at the level of the thyroid cartilage and thrombosed internal jugular vein. After meticulous removal of fishbone, a 3 × 3 mm laceration was found on the medial aspect of the internal jugular vein. Systemic heparinization was done just before ligation of internal jugular vein with a stat dose of 5000 unit unfractionated heparin administered intravenously then internal jugular vein was ligated proximal and distal to the site of injury. Perforation of the pharynx was repaired in two layers. Drainage of abscess cavities and debridement of necrotic tissues were done, and samples were sent for culture. A Penrose drain was inserted in paravertebral region, the skin was closed with separate stitches, and systemic heparinization was ceased. The postoperative course was uneventful, and liquid diet was started on the 4th postoperative day. In such settings, ultrasound and barium esophagram are useful tools to evaluate postoperative condition of the patients. Since serious intracranial sequelae after unilateral ligation of internal jugular vein are extremely rare, routine ultrasound evaluation after surgery is not advised unless signs of intracranial hypertension occur; furthermore, preoperative CT scan of the patient had documented patency of contralateral internal jugular vein, therefore after a normal barium esophagram, the patient was discharged on the 5th postoperative day. |
A 27-year-old male presented to emergency with pain and swelling on posterolateral aspect of the right knee following a varus thrust while playing cricket. Clinical examination confirmed the findings along with foot drop and dense hypoesthesia in CPN distribution. Radiological examination revealed a displaced avulsion fracture of fibular head ( and ). Magnetic resonance imaging of the right knee showed avulsion fracture of the fibular head with attached lateral collateral ligament and midsubstance tear in the posterolateral capsule of the knee along with edematous soft tissue engulfing the CPN suggestive of its compression. There was edema present in the midsubstance region of both cruciate ligaments (). There was no sign of meniscal injury. The patient was advised to undergo open reduction and internal fixation of bony avulsion from the fibular head to restore the posterolateral stability of the knee joint along with simultaneous exploration of the CPN. The right knee was examined under anesthesia, and there was Grade II opening on varus stress testing at 30° and 60° flexion. There was no other sign of instability at the right knee.\nThe right knee was approached through the posterolateral approach. The bony avulsed fragment from the fibular head with attached lateral collateral ligament and popliteofibular ligament was identified. The CPN was found to be lacerated approximately by 50% of the total diameter (). The fibular head avulsion was anatomically reduced and fixed with a single 4 mm partially threaded screw (). The knee was found to be stable after fixation. Neurolysis of CPN was done microscopically, and repair of nerve fascicles was done without tension. Post-operative bracing of the knee with intermittent range of motion was started on the 3rd day. The patient was followed up for knee stability and CPN recovery. After 1 year, post-operative knee is stable with grade 3/5 power (MRC grading) at the right ankle, and sensations recovered up to 50% over the right foot. The strengthening exercises for quadricep and hamstring group of muscles were also started in the immediate post-operative period. There was gradual improvement in sensory and motor power during the follow-up. |
A 24-year-old female patient presented with acute paraplegia which occurred immediately after a cesarean delivery under spinal regional anesthesia. She was a healthy woman without any neurological dysfunction or any other medical history. She had no important bleeding risk factors, including medical treatment for anticoagulation, and no history of heavy lifting or trauma. In another hospital, she underwent a normal cesarean section under spinal epidural anesthesia and the whole procedure had no specific complications. However, one day after delivery, paraplegia and sensory loss below D7 dermatome did not recover. In addition, she could not feel any sense of defecation and urination. Anal tone was impaired and fecal compaction was discovered. Other symptoms included a mild fever and neck stiffness as well. She was admitted to our neurosurgical department and spine magnetic resonance image (MRI) scan was performed. Her initial spine MRI scan showed an abnormal spinal cord lesion from C2 level to D5, which was suspicious of a spinal cord swelling with a proximal syrinx or a hemorrhage formation. The lesion showed heterogeneous high signal intensity on T2-weighted MRI. On T1-weighted image, the lesion showed irregular rim enhancement (). In our case, the margin of enhancing lesion was poorly demarcated. Considering the patient's history, namely, the lack of symptoms prior to delivery and then the occurrence of acute neurological symptom immediately after delivery under spinal anesthesia, acute bleeding of intramedullary tumor or infection, or other autoimmune diseases were suspicious as a differential diagnosis. The patient had a mild fever with neck stiffness, however Kernig sign and Bruzinski sign were both negative in this patient. For exclusion of central nervous system infection, cerebrospinal fluid (CSF) study and laboratory study were undertaken. On CSF study, the value of white blood cell (WBC), red blood cell, protein, and glucose was 2, 0, 37, and 82, respectively. This result was within the normal range. Additionally, CSF culture was done, however it showed no growth of bacteria. On laboratory study, WBC, erythrocyte sedimentation rate, and C-reactive protein were within normal range, however the percentage of segmented neutrophil count was increased up to 86.9%. The patient's brain MRI scan was also done and no specific abnormal lesion was observed. Antibiotics and steroid therapy were alternatively prescribed each for few days to rule out the differential diagnoses. After 2 weeks of medical treatment, the patient showed neither specific improvement nor aggravation. Then, a spine MRI scan was performed to follow up the abnormal lesion and to prepare for operation (). The patient's MRI scan showed that the lesion slightly diminished in size. To confirm the intradural lesion, wide unilateral hemilaminectomies were performed on the left side of C7 and T1 level (). After opening the dura, a midline pial incision was done precisely and a gray and sticky mass was observed right below the pial incision. After the mass was partially resected, a dark hematoma was observed inside and beneath the mass. A large amount of dark hematoma could be sucked out easily. There were no specific complications during surgery. After surgery, the patient's symptoms slightly improved. She could sense pain and temperature above the knee level. Her postoperative MRI scan showed that the lesion significantly decreased (). 5 days after surgery, a histopathological examination confirmed the diagnosis of ependymoma (World Health Organization grade II) with hemorrhagic component. On immunohistochemistry study, glial fibrillary acidic protein was weakly positive, epithelial membrane antigen was negative and Ki-67 was 4%. To entirely remove the ependymoma, we planned the second surgery one week after the first surgery. Her preoperative spinal MRI scan for the second surgery showed that a huge amount of the abnormal lesion decreased. The central portion, which was the hematoma, significantly diminished and the enhancing portion, which was an ependymoma, was partially removed as well (). Unilateral hemilaminectomies were extended from C6 level to T2 level. Although the mass was very sticky and a severe adhesion was noted, the tumor could be subtotally removed with minimal damage to the spinal cord. Interaoperative monitoring was performed and it did not show significant changes during surgery. After the second surgery, the patient's postoperative spinal MRI scan showed that most of the enhancing mass was removed and that only a very thin layered enhancing portion remained at C7 level (). In conclusion, the tumor was subtotally removed. The patient's sense of temperature improved above the thigh level. However, her motor functions and cauda equine syndrome did not improve. Her Karnofsky performance scale score was 50. The patient was under the rehabilitation therapy and then was discharged without any other complications. At present, radio-therapy for the remaining lesion is planned, according to previous reports which insisted benefits in progression free survival of adjuvant radiotherapy in the patients with spinal ependymoma []. |
A 28 year-old male was found to have a slightly increased serum creatinine (1.7 mg/dl) during the admission to ER because of abdominal pain. Since both his past medical history and an abdominal ultrasound performed on admission were unremarkable he was discharged with the indication to see a Nephrologist. At the Nephrology outpatient clinic, because of the reduced eGFR and of the presence of proteinuria (300 mg/24 h), an hospitalization for further investigations was planned.\nWhile waiting for the admission, a couple of weeks later, the patient was admitted to the emergency room in another hospital for abdominal pain and dyspnea. On admittance the ECG tracing performed showed sinus rhythm with diffuse repolarization abnormalities. Further exams revealed an increase of troponin (54 ng/ml) and transaminases levels (GOT 50 U/L; GPT 125 U/L) and a cardiac ultrasound showed a severe left ventricular dysfunction with right heart failure. Patient was thus admitted to the coronary intensive care unit. During his hospital stay the possibility of acute myocarditis was excluded because of the absence of a recent viral syndrome and the negativity of IgM antibodies against the viruses most commonly affecting the cardiovascular system and because of the low inflammatory indexes. An abdominal ultrasound demonstrated abdominal and pleural effusions, regular size and morphology of spleen and liver, no pancreatic changes; both kidneys had normal size but showed irregular margins and a reduced thickness of the cortex, no hydronephrosis nor stones were observed. A magnetic resonance demonstrated an increased volume of the cardiac chambers with a severe reduction of the function of both ventricles (EF20%), however no signs of reduced myocardial perfusion, valvular defects or other tissue changes such as inflammation, fibrosis or edema could be demonstrated. A chest CT scan confirmed the presence of pleural effusion and showed parenchymal atelectasias mainly involving the lower lobes of the lungs. The patient was discharged after 11 days on diuretics (furosemide 100 mg/daily), B-blocker (carvedilol 37.5 mg/daily), acetyl salicylic acid (Aspirin 100 mg/day) and an Ace inhibitor (Ramipril 5 mg/day) with a diagnosis of dilated cardiomyopathy and severe left ventricular dysfunction complicated with cardiogenic shock and anasarca. Few days later an EKG ergometric test revealed no areas of inducible ischemia. A further admission to the Cardiology Unit to define the effect of treatment was performed a week later. Cardiac ultrasound confirmed the left ventricular dysfunction (EF 23%), a coronary angiography was negative and a right chamber catheterism and endomyocardic biopsy demonstrated no specific lesion nor inflammatory infiltrate. The patient was then admitted to our Nephrology Unit, as planned, to evaluate the cause of the chronic kidney disease. On admission the patient appeared healthy and well oriented, his blood pressure was 110/60 mmHg, heart rate 56 bpm, physical examination was unremarkable except for a 1/6 intensity systolic heart murmur better audible on mitral valve area. ECG tracing showed sinus bradycardia. Laboratory studies revealed an increased serum creatinine (1.87 mg/dl), eGFR 48 ml/min/1.73 m2, hypercalcemia (serum calcium 11.5 mg/dl), hemoglobin 15.1 g/dl, sodium 135 mEq/L, potassium 4.37 mEq/L, phosphate 3.1 mg/dl, serum albumin 4.07 g/dl. The patient denied use of vitamin D analogs thus in consideration of the unexplained hypercalcemia other exams were performed. Thyroid hormones, calcitonin, vitamin D, serum ACE, phosphate and calcium urinary excretion rate were all within the normal range. PTH was lower than 3 pg/ml. To rule out multiple myeloma serum protein electrophoresis, serum and urine immunofixation, and spine and skull x-ray were performed and did not show any pathological changes.\nRenal biopsy was finally performed without complications and the patient was discharged after 24 h. While waiting for the renal biopsy pathology report, medications were not modified, but for an increase of the dose of furosemide because of the hypercalcemia. The renal biopsy contained eight glomeruli none of which was globally or partially sclerotic and no pathological changes were observed at glomerular level, vessels were also unremarkable. Tubulointerstitium was the only affected compartment with a moderate to severe inflammatory infiltrate, some calcium crystals in the tubular lumen and two granulomas with multinucleated giant cells (Figs. and ). No necrosis could be observed within the granulomas. A diagnosis of granulomatous interstitial nephritis due to sarcoidosis was made.\nWhen the patient was called to discuss his kidney biopsy, his serum creatinine had further increased (3.5 mg /dl), eGFR 22 ml/min/1.73 m2 and so had his serum calcium (13.3 mg/dl), serum albumin 4.06 g/dl. For this reason, he was hospitalized again and treated with pulse methylprednisolone (250 mg/day for three days) and then with maintenance dose prednisone (50 mg/day). Serum creatinine decreased in a few days and so did his serum calcium. Since the episode of acute kidney injury the patient has been followed at our unit and at the unit of Cardiology. Renal function returned to normal (serum creatinine 1.1 mg/dl, eGFR 91 ml/min/1.73 m2) and heart function markedly improved over the following two years (EF 49% vs 37% vs 32% vs 23%). The diuretic and cardiologic therapy was significantly reduced to only 25 mg of furosemide and a small dose of beta blocker. The steroid was gradually tapered to 5 mg/day of prednisone for one year and then stopped. |
A 72-year-old male with known myelodysplastic syndrome presented to of Seoul St. Mary’s Hospital with a 7-day history of fever and dyspnea. His planned chemotherapy for myelodysplastic syndrome had been stopped a month before due to fungal infection of the orbit. The initial absolute neutrophil count was only 130/mm3, and antibiotic therapy was initiated under the possibility of pneumonia or aggravation of orbital infection. Even after 7 days of antibiotic therapy, the fever persisted and no evidence of infection was found. We performed transthoracic echocardiography under suspicion of infective endocarditis; a 13 mm echogenic mass was found on the atrial side of the tricuspid valve (). Similar findings were also revealed by transesophageal echocardiography. Fortunately, no functional abnormalities of the valve were seen on echocardiography.\nBased on the clinical symptoms and the echocardiographic findings, the patient was highly suspected of having infective endocarditis of the tricuspid valve. He was scheduled for emergency surgical removal due to the possibility of fungal endocarditis and the poor clinical course of infective endocarditis in immunocompromised patients. With a cardiopulmonary bypass, the right atrium was opened after aortic cross-clamping and cardioplegic arrest. On the exploration, a solitary polypoid gelatinous mass 13×16 mm in size infiltrating the septal leaflet of the tricuspid valve was found in the surgical field (). Although the tumor had a stalk, simple resection without making a defect in the septal leaflet appeared to be impossible, because the leaflet was too thin. Therefore, the mass was excised with part of the septal leaflet of the tricuspid valve using the quadrangular resection technique, followed by reconstruction with autologous pericardium. After successful weaning from the cardiopulmonary bypass, intraoperative transesophageal echocardiography was performed, showing no residual tumors. The tricuspid valve was found to be competent without any functional deficits after operation. Papillary fibroelastoma was pathologically confirmed, and no evidence of infective endocarditis was found (). Follow-up transthoracic echocardiography showed no residual or recurrent tumor, with uneventful postoperative recovery. |
A 73-year-old man was referred from a local hospital with impaired consciousness and left hemiparesis after a motor car accident. He was diagnosed with traumatic subarachnoid hemorrhage on the left parietal lobe and left lateral mass fractures on C2, C5, and C6, involving the transverse foramen (). A neurological examination revealed stupor with a Glasgow Coma Scale score of 8. Severe grade 2 hemiparesis of the left arm and leg was detected. High signal was seen at the C6-7 levels of the spinal cord on a sagittal cervical spine magnetic resonance imaging (MRI). The signal void on the left vertebral artery was lost on the C6 axial image (). He was diagnosed with left VAD and was managed conservatively. Neck computed tomography (CT) angiography showed obliteration of the left second segment of the vertebral artery (V2) (). The obliteration of the V2 segment remained on 2-week follow-up CT angiography. A brain MRI was performed to evaluate the cause for impaired consciousness and motor weakness. Diffusion weighted images revealed multiple area of abnormally high signals in the right side of the pons, left side of the midbrain, right thalamus, corpus callosum splenium, both parietal lobes, and the left frontal lobe, indicating multiple acute cerebral infarctions (). Ischemic areas were diffusely distributed to the anterior and posterior circulation of the Circle of Willis. Ischemic stroke occurred immediately after cervical spine trauma and neurological deficits after trauma resulted from brain ischemia. Aspirin was started for antiplatelet therapy 1 week after the trauma after confirming disappearance of the acute traumatic hemorrhage. He was discharged from the hospital on aspirin to receive neurological follow-up and rehabilitation therapy. A brain CT scan at discharge showed subdural fluid collection in both frontal lobes. His motor weakness and consciousness state were improving gradually to a grade 4 weakness and alert state on follow-up examination. Bilateral chronic subdural hematomas (CSDHs) were observed with headache 4 months after the trauma (). Burr hole trephinations were performed on both sides for the CSDHs. |
A 54-year-old Japanese woman visited our ophthalmology department after experiencing proptosis, lid swelling, diplopia, and retro-orbital pain in her left eye lasting for 1 day. She had a medical history of poorly differentiated adenocarcinoma of the stomach, which had metastasized to her ovary and mesentery, diagnosed 2 years earlier. She had undergone four regimen courses of chemotherapy, yet these had failed and she thus received palliative treatment. There were metastases to subcutaneous tissue of her neck and thoracic bone marrow 3 months before her initial visit to our ophthalmic department. She had been admitted to our hospital 5 days previously without symptoms in either eye. She had undergone stenting in her esophagus against eating difficulties but she lived a self-reliant life at home.\nAt her first visit, an external examination showed lid swelling, red coloration, and proptosis of her left eye. A motility examination revealed an adduction deficit of − 4.0 and an abduction deficit of − 1.0. Ophthalmological examinations revealed a best-corrected visual acuity of 20/20 and an intraocular pressure of 15 mmHg in both eyes. No abnormal findings were found in the anterior segment. Her pupils were equally reactive without any relative afferent pupillary defect. A funduscopic examination showed partial optic disc edema in her left eye (Fig. a). No choroidal masses or striae were noted.\nA CT scan performed 10 days before her initial visit to our ophthalmology department revealed enlargement of the left medial rectus muscle. Retrospectively, similar findings were seen on a CT scan performed 3 months previously, and had worsened in the interim. Yet, a CT scan that had been performed 6 months previously showed no remarkable findings (Fig. ). There was no enlargement of other extraocular muscles and no swelling or mass lesion in other orbital tissues during the 6 months. So, gastric cancer metastasis to the medial rectus muscle of her left eye was suspected. Radiation therapy for metastasis to the subcutaneous tissue of her neck and thoracic bone marrow was effective; she received a total of 20 Gy/5 courses of radiation therapy to the orbit. A few days after completion of radiation therapy, lid swelling, red coloring, and pain disappeared. Two weeks post-radiation therapy, a motility examination revealed an adduction deficit of − 4.0 and Hertel’s exophthalmometry measurements with a 108-mm base were 14 mm (right eye) and 19 mm (left eye). At 1.5 months post-radiation therapy, a motility examination revealed an adduction deficit of − 2.0 and Hertel’s exophthalmometry measurements (108-mm base) were 14 mm (right eye) and 13 mm (left eye). A posterior ocular segment examination showed a normal left optic disc (Fig. b). She died 3 months after her initial presentation to our ophthalmology department. |
A 28 year-old male was found to have a slightly increased serum creatinine (1.7 mg/dl) during the admission to ER because of abdominal pain. Since both his past medical history and an abdominal ultrasound performed on admission were unremarkable he was discharged with the indication to see a Nephrologist. At the Nephrology outpatient clinic, because of the reduced eGFR and of the presence of proteinuria (300 mg/24 h), an hospitalization for further investigations was planned.\nWhile waiting for the admission, a couple of weeks later, the patient was admitted to the emergency room in another hospital for abdominal pain and dyspnea. On admittance the ECG tracing performed showed sinus rhythm with diffuse repolarization abnormalities. Further exams revealed an increase of troponin (54 ng/ml) and transaminases levels (GOT 50 U/L; GPT 125 U/L) and a cardiac ultrasound showed a severe left ventricular dysfunction with right heart failure. Patient was thus admitted to the coronary intensive care unit. During his hospital stay the possibility of acute myocarditis was excluded because of the absence of a recent viral syndrome and the negativity of IgM antibodies against the viruses most commonly affecting the cardiovascular system and because of the low inflammatory indexes. An abdominal ultrasound demonstrated abdominal and pleural effusions, regular size and morphology of spleen and liver, no pancreatic changes; both kidneys had normal size but showed irregular margins and a reduced thickness of the cortex, no hydronephrosis nor stones were observed. A magnetic resonance demonstrated an increased volume of the cardiac chambers with a severe reduction of the function of both ventricles (EF20%), however no signs of reduced myocardial perfusion, valvular defects or other tissue changes such as inflammation, fibrosis or edema could be demonstrated. A chest CT scan confirmed the presence of pleural effusion and showed parenchymal atelectasias mainly involving the lower lobes of the lungs. The patient was discharged after 11 days on diuretics (furosemide 100 mg/daily), B-blocker (carvedilol 37.5 mg/daily), acetyl salicylic acid (Aspirin 100 mg/day) and an Ace inhibitor (Ramipril 5 mg/day) with a diagnosis of dilated cardiomyopathy and severe left ventricular dysfunction complicated with cardiogenic shock and anasarca. Few days later an EKG ergometric test revealed no areas of inducible ischemia. A further admission to the Cardiology Unit to define the effect of treatment was performed a week later. Cardiac ultrasound confirmed the left ventricular dysfunction (EF 23%), a coronary angiography was negative and a right chamber catheterism and endomyocardic biopsy demonstrated no specific lesion nor inflammatory infiltrate. The patient was then admitted to our Nephrology Unit, as planned, to evaluate the cause of the chronic kidney disease. On admission the patient appeared healthy and well oriented, his blood pressure was 110/60 mmHg, heart rate 56 bpm, physical examination was unremarkable except for a 1/6 intensity systolic heart murmur better audible on mitral valve area. ECG tracing showed sinus bradycardia. Laboratory studies revealed an increased serum creatinine (1.87 mg/dl), eGFR 48 ml/min/1.73 m2, hypercalcemia (serum calcium 11.5 mg/dl), hemoglobin 15.1 g/dl, sodium 135 mEq/L, potassium 4.37 mEq/L, phosphate 3.1 mg/dl, serum albumin 4.07 g/dl. The patient denied use of vitamin D analogs thus in consideration of the unexplained hypercalcemia other exams were performed. Thyroid hormones, calcitonin, vitamin D, serum ACE, phosphate and calcium urinary excretion rate were all within the normal range. PTH was lower than 3 pg/ml. To rule out multiple myeloma serum protein electrophoresis, serum and urine immunofixation, and spine and skull x-ray were performed and did not show any pathological changes.\nRenal biopsy was finally performed without complications and the patient was discharged after 24 h. While waiting for the renal biopsy pathology report, medications were not modified, but for an increase of the dose of furosemide because of the hypercalcemia. The renal biopsy contained eight glomeruli none of which was globally or partially sclerotic and no pathological changes were observed at glomerular level, vessels were also unremarkable. Tubulointerstitium was the only affected compartment with a moderate to severe inflammatory infiltrate, some calcium crystals in the tubular lumen and two granulomas with multinucleated giant cells (Figs. and ). No necrosis could be observed within the granulomas. A diagnosis of granulomatous interstitial nephritis due to sarcoidosis was made.\nWhen the patient was called to discuss his kidney biopsy, his serum creatinine had further increased (3.5 mg /dl), eGFR 22 ml/min/1.73 m2 and so had his serum calcium (13.3 mg/dl), serum albumin 4.06 g/dl. For this reason, he was hospitalized again and treated with pulse methylprednisolone (250 mg/day for three days) and then with maintenance dose prednisone (50 mg/day). Serum creatinine decreased in a few days and so did his serum calcium. Since the episode of acute kidney injury the patient has been followed at our unit and at the unit of Cardiology. Renal function returned to normal (serum creatinine 1.1 mg/dl, eGFR 91 ml/min/1.73 m2) and heart function markedly improved over the following two years (EF 49% vs 37% vs 32% vs 23%). The diuretic and cardiologic therapy was significantly reduced to only 25 mg of furosemide and a small dose of beta blocker. The steroid was gradually tapered to 5 mg/day of prednisone for one year and then stopped. |
The patient was 52-year-old Caucasian male, with no significant past medical history, who presented for routine follow-up after having been diagnosed with carcinosarcoma of the right parotid gland with a metastatic deposit within the jugular foramen, 18 months after diagnosis and 1 year after completion of treatment. He was an avid smoker of 1 to 1.5 packs per day, did not have a family history of cancer, and had no environmental exposures during work.\nAt the time of diagnosis, 18 months prior, initial pathological and histological analysis displayed a malignant neoplasm of the parotid gland with a biphasic histological pattern. The minority component had overtly glandular epithelial features characterized by well-defined islands of neoplastic cells. These cells were positive for pankeratin and epithelial membrane antigen. Special stains also revealed intraluminal/intracystic mucin. Surrounding the aforementioned neoplastic islands were cells showing a solid growth pattern. These cells were negative for pankeratin and only a small number showed staining for epithelial membrane antigen.\nAfter discussion and presentation at tumor board the patient had started an aggressive treatment regimen that included a radical parotidectomy, complete with neck dissection and excision of the jugular foramen mass using gamma knife radiosurgery. Given the aggressiveness of the disease, on completion the patient was referred to medical oncology for concurrent chemoradiation. At that time the patient received 3 cycles of cisplatin chemotherapy at a dosage of 100 mg/m2 along with X-ray radiation therapy. The positron emission tomography (PET) scan following completion of treatment showed no evidence of local recurrence within the parotid or mastoid region. Furthermore, there was no radiological evidence of distal recurrence using flurodeoxy glucose–labeled PET scanning.\nAt this time the patient had then presented to the clinic for a routine 6-month follow-up visit (a year after completion of therapy) with no complaints or symptoms. The oral cavity and oropharyngeal examination revealed no lesions on presentation. Both ear canals were patent and the patient’s tympanic membranes were clear. Visual acuity was unchanged and hearing was intact. Facial sensation was intact; however, the patient showed absent motor response on the right side, which had presented after initial therapy and was since unchanged. Palate elevation as well as shoulder strength was symmetric, the tongue was mobile bilaterally, and no other abnormalities were noted on a complete examination. The patient’s vital signs were stable and within normal limits with temperature of 36.1°C, blood pressure of 124/77 mm Hg, a pulse of 74, and a respiratory rate of 16.\nRepeat PET and computed tomography (CT) scan were done following patient’s clinic visit, which had displayed widespread bone metastases. Multiple metastases in the spine were of concern regarding pathologic fracture potential, especially at C1 (as seen in ). There were new areas of hemorrhage in the brain with surrounding edema that were noted as well (as seen in ). Areas including the posterior left occipital lobe and posterior parasagittal left parietal lobe showed hemorrhagic metastases although the patient remained asymptomatic.\nThe magnetic resonance imaging of the brain also showed significant metastatic activity. There was a lobulated enhancing intra-axial mass lesion within the left precuneus that measures 2.7 cm × 1.6 cm × 1.8 cm, and a large enhancing lobulated mass lesion occupying a large portion of the left occipital lobe that measured approximately 2.5 cm × 4.1 cm × 3.8 cm. There was vasogenic edema around larger lesions located within the left precuneus and left occipital lobe with resultant mass effect on the atrium (as seen in ).\nThere has been marked progression of widespread bone metastases throughout the spinal column including but not limited to the following new metastases at C4, right lamina of T1 with lytic destruction extending into the spinal canal, lytic left anterior T4, including left fifth rib posterolaterally. Additional lytic metastasis were seen on the left section of T8 with a larger and a much more intense, right transverse process of T9, left T11, and at the right anterior region of T12. However, there was no recurrent tumor seen in the right parotid bed.\nOn repeat biopsy a well to moderately differentiated adenocarcinoma with strikingly cribriform growth pattern and large areas of necrosis was seen. The tumor morphology was similar to that of the carcinomatous portions of the previously diagnosed carcinosarcoma of parotid, indicating that the metastatic brain tumor likely represented a metastatic salivary ductal adenocarcinoma component of carcinosarcoma.\nThe cancer therefore recurred in this asymptomatic patient nearly a year since completion of aggressive treatment, which consisted of a combination of surgery, gamma knife surgery, chemotherapy, and radiation. Unfortunately, with the dramatic increase in tumor burden, as well as the patient not wanting further aggressive treatment, the patient opted for hospice care and peacefully succumbed to his disease. |
Ms CJ was a 76-year-old lady who presented with vaginal pain and discharge. She had a past medical history of asthma, hypertension, hyperlipidaemia, type II diabetes mellitus, and congenital right hydronephrosis (due to a congenital vascular abnormality, treated with a ureteric stent). She had a left salpingo-oophorectomy for a nonmalignant lesion of the ovary.\nOn examination she was noted to have a 4 cm mass in the vagina and biopsy of this confirmed melanoma of a balloon type. Preoperatively CT scan did not show any evidence of distant metastases and an MRI scan of the pelvis demonstrated the lesion, but no associated lymphadenopathy (T4N0M0). She was discussed at the regional tumour-board meeting and considered for neoadjuvant treatment; however no appropriate systemic therapy was available. She therefore underwent extensive surgery consisting of an anterior exenteration in which her bladder, uterus, and remaining right ovary were removed. Intraoperative concerns regarding attaining clear margins lead to the decision to perform vaginectomy also. Tumour was resected completely and Ms CJ made a good recovery and experienced no perioperative complications. Histology confirmed a urethral nodular malignant melanoma. Further discussion was performed at the melanoma tumour-board meeting with the decision of close follow-up. Gynaecological oncology clinic review at 3 weeks and 12 weeks postoperatively revealed no complications.\nMacroscopic histopathological examination revealed a pelvic exenteration specimen measuring 140 × 135 × 62 mm. It comprised vagina measuring 66 × 49 × 25 mm which contained a pigmented and nodular tumour measuring 28 × 25 × 21 mm which lay 20 mm from the distal resection margin. The tumour was situated on the anterior wall of the vagina. The specimen also contained a bladder measuring 40 × 38 × 36 mm, the mucosa of which appeared normal. There was attached urethra measuring up to 30 mm in length which appeared close to the tumour distally.\nMicroscopic examination showed a tumour arising from the urethra which showed squamous metaplasia. The lesion was nodular and was composed of cytologically atypical cells with pigmentation in areas. The cells were pleomorphic and hyperchromatic and some cells showed prominent nucleoli. There were epithelioid areas, along with areas showing cleared cytoplasm and elsewhere the tumour displayed a spindle cell architecture. There were occasional atypical multinucleated cells. Overall the features were of a nodular malignant melanoma. The lesion measured 22 mm in thickness and there was evidence of ulceration within the urethral epithelium. Six mitotic figures were identified per 10 high power fields. There were no lymph node invasion identified and no lymphovascular, perineural invasion or microsatellites. The tumour abutted the circumferential margin but did not infiltrate into it and lay 19 mm from the distal resection margin. |
Our proband is a 43-year-old male of northern European descent who initially presented with acute onset left leg cramping pain that lasted for several days. The pain was associated with numbness in his left leg and nausea. His past medical history was significant for mitral valve prolapse, aortic root dilation umbilical hernia left anterior cruciate ligament (ACL) rupture, and severe myopia corrected with LASIK surgery. Family history revealed rheumatic heart disease in his mother, necessitating mitral valve repair due to mitral valve prolapse with severe regurgitation. Upon examination, his popliteal arterial pulses were 4+ bilateral, bounding. His height was 196 cm, with an increased arm span-height ratio of 1.07. He had a mild pectus carinatum deformity, mild scoliosis positive wrist and thumb sign, pes planus, and mild skin striae. A Doppler ultrasound was performed by his primary care physician (PCP), which ruled out deep venous thrombosis (DVT); however, it revealed a large left popliteal artery aneurysm with thrombus that measured 6.3 × 3.1 × 3.4 cm. He underwent a CT angiogram that showed normal abdominal aorta as well as common iliac arteries. However it showed bilateral popliteal artery aneurysms measuring 3.6 × 4 cm in the left side and 3 × 2.8 cm in the right side (). After repairing of his left popliteal artery aneurysm with bovine carotid interposition via posterior approach, he was sent for cardiac and genetic evaluations. He underwent a MRI exam of the heart with and without intravenous gadolinium contrast administration () which showed mild bileaflet mitral valve prolapse and aortic root dilation (measuring 46 mm at the level of the sinuses of Valsalva). The patient has been prescribed metoprolol tartrate and losartan to control his aortic root disease and was to undergo elective aortic repair when the aortic dimension becomes near to or reaches 50 mm. A diagnosis of Marfan syndrome was made based on the revised Ghent criteria as the proband had aortic root dilation (Z-score: 3.72) and a systemic score of 12 (). His diagnosis was further confirmed after undergoing FBN1 sequencing and deletion/duplication analysis, which detected a novel pathogenic variant in gene FBN1, denoted by c.5872 T>A (p.Cys1958Ser) (). |
Patient 2 is a 19-year-old male adolescent that developed his first motor tics at the age of eight. No other comorbidities were present, and he had a negative family history for tic disorders. Tics started with head shaking, followed by eye blinking and grimaces, and later progressed to his shoulders, arms, trunk and legs. The most debilitating tics were persistent bouts of motor tics in his entire body. He also experienced mild vocal tics like coughing, sniffing and grumbling. At the age of 18, he had to quit school since he was not able to concentrate anymore due to the severity of the tics and because his tics were an unacceptable distraction to fellow students. Medication and behavioural therapy had not been effective in the past. He had used clonidine, pimozide, haloperidol and quetiapine either without lasting positive effects or with intolerable side effects. Between 2007 and 2009 he underwent several psychotherapeutic therapies, among which exposure and response prevention, habit reversal and relaxation training. He was using cannabis daily since he felt it calmed him. This patient fulfilled our selection criteria (Table ) and was indicated for bilateral DBS of the anterior GPi. He agreed to stop using cannabis as part of the treatment agreement, so that better evaluation of symptoms during programming would become possible. The surgery was carried out without complications in March 2016 (at the age of 19).\nWithin 1 week after the surgery the stimulator was turned on and systematically different stimulation parameters were tested. Initially he experienced increased agitation, but also an increase in tic frequency and severity. Despite he was informed that finding the correct stimulation parameters would take time, he had hoped for an immediate effect on his tics and was disappointed. This resulted in tension, anger and arguments with his supporting parents, making the programming sessions more difficult. In the following weeks, we had several programming sessions at the outpatient department, mainly decreasing the voltage and slowly increasing it at home, and finally with a voltage of 3.0 a significant tic reduction was established and the agitated feeling had almost disappeared. However, at home the situation escalated, mainly due to the daily excessive use of cannabis, with increasingly quarrelsome and aggressive behaviour. He still lived at home with his parents and siblings who tried to support him and this lead to severe conflicts. The exact reason why he started using drugs again is uncertain, but he told us he was overwhelmed, felt a lot of pressure to succeed with the therapy, and needed the cannabis to calm down, even though he knew it caused him more problems. We offered psychological treatment, which he rejected.\nIn July 2016, we arranged a meeting with the Committee on Medical Ethical Issues of our hospital to discuss how to proceed: we were not able to reliably evaluate the effects of the DBS due to the excessive use of cannabis and non-compliance of the patient and doubted whether continued treatment was useful. Also, the potential contribution of the DBS to the aggressive behaviour was discussed. He was known with aggressive behaviour following cannabis use and it seemed unlikely that the DBS settings were the cause of his behavioural problems. Therefore, we decided to continue treatment because of its positive effect on tics. However, we discussed with the patient that we would not further adjust the stimulation settings if he would not be compliant with all aspects of the treatment agreement, including stopping cannabis usage. A few months later, his behaviour drastically changed in a positive way. He stopped smoking cannabis, his aggressive behaviour as well as the relationship with his parents improved, and he found a job and started working again. One year after surgery, he was quite satisfied with the effects of DBS. All tics were still present, but much less frequent and intense and he could suppress them better. He did not suffer from side-effects anymore. At that moment, his YGTSS was reduced from 32 preoperatively (motor/vocal = 23/9) to 10 postoperatively (motor/vocal = 8/2). |
A 24-year-old male presented with neck pain following road traffic accident four months ago. He had had a head-on collision with an oil tanker while he was in the backseat of the car. The probable mechanism of injury was postulated to have been flexion distraction injury at the sub axial spine. He experienced severe no radiating pain in the neck. There was no history of loss of consciousness, seizure, vomiting, or bleeding through the ear, nose, or throat. He also complained of tingling sensations in bilateral upper limbs; however, there were neither muscular weaknesses nor gait disturbance. There were no bladder or bowel complaints. No steroidal anti-inflammatory drugs (NSAIDs) were prescribed by a general physician for neck pain and no radiographic examination of the neck was performed. There was a decrease in intensity of neck pain and tingling sensations since the time of injury. The patient referred to our out-patient department four months after the injury complaing of neck pain, which was mild to moderate in intensity. The physical examination revealed restriction of neck movements. Neurological examination revealed no significant finding for tone and power in all the four extremities. There was no sensory disturbance and deep tendon reflexes examination findings were unremarkable. The radiographs of the neck suggested subluxation of the fifth cervical vertebrae (C5) over the sixth cervical vertebrae (C6) with greater than 50% translocation in the anteroposterior plane and interspinous widening (). Computed tomography (CT) revealed bilateral facet dislocation of C5 over C6 (). Magnetic resonance imaging (MRI) confirmed the CT findings of bilateral facet dislocation and compression of the cervical cord with disc prolapse at the C5- C6 level (). Closed reduction of the dislocation was not attempted as four months had passed since the time of injury and there were signs of partial facet fusion on CT.\nA 35-year-old male fell from a height of approximately ten feet and experienced pain in the neck along with numbness in bilateral upper limbs. There was no motor weakness of the limbs and no bladder or bowel problems. A local physician had prescribed NSAIDs for his neck pain. No radiographic examination was performed. He continued to experience neck pain with intermittent relief after taking NSAIDs. Five months after the injury, he referred to our institute complaining of neck pain. The pain was nonradiating but there was numbness in both upper limbs since the injury. On examination, there was restriction of neck movements. Clinical examination was otherwise unremarkable.The X-ray, CT, and MRI of the neck revealed bilateral facet dislocation at C5-C6 (). Closed reduction was not attempted in this case, due to the long time lapse since injury and partial fusion at the C5-C6 vertebrae.\nThe patients were operated by the senior author (KF) with the patient in supine position on a radiolucent table with Crutchfield tongs in situ. The cervical spine was exposed by standard anterior approach. The fibrocartilagenous tissue and bone were removed from the anterior surface of the C5 and C6 vertebral bodies. A complete microscopic anterior discectomy was performed at C5-C6 level. Temporary wound closure of skin and subcutaneous tissue was done.\nThe patient was then turned to the prone position and a standard posterior midline exposure was performed from C3 to C7. Bilateral inferior facets of the superior vertebrae were perched over the superior articulating facets of the inferior vertebrae. There was partial bony fusion between locked facets. The superior articulating facets of the inferior vertebrae were partially resected to achieve reduction. Lateral mass screws at C5 and C6 vertebra were inserted under fluoroscopic guidance. The reduction was achieved with the help of progressive extension. The reduction was confirmed under fluoroscopy. The rods were secured on the screws and the wound was closed in layers. Then the patient was carefully turned to supine position for instrumented anterior cervical interbody fusion. The anterior wound was reopened. The interbody area was prepared and measured with a calibrated device and a tricortical iliac crest bone graft of the same dimensions was harvested. The graft was inserted in the interbody area and secured with a locking plate (AO, Synthes, Solothurn, Switzerland). The wound was closed with a suction drain. The duration of surgery was about 240 minutes with blood loss of about 350 mL.\nPostoperatively, the patients were mobilized in a Philadelphia collar for eight weeks. There was no postoperative neurological deficit and wound healing was uneventful. Follow-up radiographs included anteroposterior, lateral, flexion, and extension views () along with CT scanning at six months (). The neck movements were also painless at six months follow-up. The clinical and radiological signs were suggestive of good interbody fusion. Both patients returned to work. |
A 55-year-old man with a 28-year history of diabetes was referred to our retina clinic for a regular ophthalmic examination for diabetic retinopathy. At the time of the first visit, the patient presented with proliferative diabetic retinopathy in both eyes and clinically significant macular edema. The patient was also receiving peritoneal dialysis for chronic renal failure. He had undergone pars plana vitrectomy for proliferative diabetic retinopathy in combination with phacoemulsification and implantation of a hydrophilic acrylic IOL in the capsular bag of the right eye at another hospital two months prior. In addition, the patient received panretinal photocoagulation. At the time of his first visit, the best corrected visual acuity (BCVA) was 0.08 in the right eye. Four months later, the BCVA of the right eye had decreased to a finger count at 10 cm and the intraocular pressure was 38 mmHg as measured by applanation tonometer. The patient was diagnosed with neovascular glaucoma accompanied by hyphema. He underwent Ahmed valve implantation through pars plana vitrectomy at our hospital immediately after the diagnosis of neovascular glaucoma. Thereafter, the corrected visual acuity of the right eye was maintained at 0.08.\nIOL opacification, causing decreased visual acuity, was found five months following the surgery for glaucoma (ten months following the implantation of the IOL) during monitoring of the clinical course (). There was a concurrent presence of microhyphema as we monitored the clinical course. However, after the disappearance of hyphema, his visual acuity of the right eye dropped to hand motion and did not improve. The IOL was explanted from the right eye 45 months after implantation. For the explantation of the lens, the fibrotic anterior capsule was released by endoscissors under viscoelastics. The haptics were separated from the optics and two of them were explanted. The other two haptics were not explanted because we were concerned about zonular dialysis. The optics were relieved from the capsular bag by manual dissection with a spatula and viscodissection. It was then cut into small pieces with DeWecker scissors. The IOL was explanted, piece by piece, through a 3.0 mm corneal incision. A hydrophobic acrylic IOL (YA-60BB; hoya Inc., Tokyo, Japan) was then implanted into the sulcus. There was a well-circumscribed centrally and paracentrally located opacification of the original IOL optic. The opacification was generalized, white, homogenous and it affected the entire surface inside of the IOL. Postoperatively, the patient did not exhibit specific complications and the visual acuity improved to 0.04. Six months after the operation, the corrected visual acuity was maintained at 0.06 due to macular edema. |
A 65-year-old female with a history significant for tobacco use, chronic obstructive pulmonary disease (COPD), tobacco abuse, sarcoidosis, hypertension, hyperlipidemia and non-insulin-dependent diabetes mellitus presented for the first time to outpatient cardiology clinic for evaluation of her chest pain. She had initially seen her primary care physician in the outpatient setting and was ruled out to have pulmonary, musculoskeletal and gastrointestinal etiology, and was subsequently referred to cardiology clinic for evaluation of cardiac etiology for her consistent chest pain symptoms despite being on medical therapy.\nThe patient reported progressive worsening of chest pain for a few months. She described radiation of the chest pain to jaw and left upper chest along with left upper extremity and shoulder area. She denied any specific association of pain with scale of activity and pain occasionally happened at rest as well. She was recently given nitroglycerin sublingual tablets for relief of her chest pain syndrome. Physical exam revealed normal cardiac findings with regular rate and rhythm. She had no murmurs and jugular venous pressure was not elevated. Electrocardiogram (EKG) showed normal sinus rhythm and left axis deviation (Figure ).\nA chest X-ray was unremarkable for any acute cardiopulmonary process (Figure ).\nAn echocardiogram was ordered, which showed normal left ventricular size with an estimated ejection fraction of 55%. She had concentric remodeling of the left ventricle. Echocardiogram was positive for akinesis of myocardial segments (Figure ).\nGiven her abnormal echocardiogram and high pre-test probability of ASCVD and symptoms concerning for unstable angina, she was offered a left heart catheterization and coronary angiography to further delineate her coronary anatomy.\nShe was brought to the catheterization laboratory in the fasting state. Coronary artery angiography and left heart catheterization were performed via right radial artery access without complications. It revealed an anomalous left main coronary artery from single right coronary ostium (Figure ).\nIt further revealed significant obstructive multi-vessel coronary artery disease involving distal left main artery, proximal left anterior descending artery, left circumflex and right coronary arteries (Figure ).\nThe patient had a right dominant system with absent left coronary cusp (Figure ).\nShe had a normal left ventricular end diastolic pressure as well. She had a high Syntax score of 34, and subsequently a Heart Team approach was pursued. She had acceptable Society of Thoracic Surgeons (STS) risk, and hence was referred for multi-vessel surgical revascularization. |
A 12-year-old female patient was referred by a private practitioner who detected a mass of soft tissue on the palatal aspect of tooth 11. The patient had a history of alveolar cleft repair done about 1 year back. Clinical examination revealed an extensive soft tissue mass in a palatal defect of tooth 11 []. Gentle exploration revealed that besides loss of the palatal enamel there was also loss of enamel from the interproximal areas of the tooth. However, the labial surface of the tooth was intact. The tooth responded to thermal and electric pulp testing within normal limits. A periapical radiograph revealed a large irregular radiolucency in the coronal region of the tooth extending mesiodistally and slightly into the coronal radicular dentin []. A diagnosis of Class 2 external cervical resorption was made.\nGlycerol was applied to the adjacent tissues to form a protective film. Isolation was achieved using the cuff rubber dam technique. A small cotton pellet was then dipped in 90 percent trichloroacetic acid and the excess was removed by dabbing it on a piece of gauze. The cotton pellet was then applied over the resorptive tissue mass with gentle pressure for about 1 minute. The tissue mass underwent necrosis with repeated application of trichloroacetic acid which was curetted out till a sound dentinal base was revealed []. Care was taken to see that the interproximal areas of the tooth were thoroughly debrided. The cavity was then refreshed with a high speed bur and restored with glass ionomer cement [].\nTrichloroacetic acid is a chemical escharotic agent that causes coagulation necrosis and renders the resorptive tissue avascular.[] The use of trichloroacetic acid does not necessitate raising a flap to gain access to the lesion. This eliminates the possibility of active resorbing cells present in the flap from being repositioned over the repaired root surface.[] However, due to the caustic nature of the acid, certain guidelines need to be followed as outlined by Heithersay:[] a. The adjacent soft tissues should be protected with glycerol, b. A thin glycerol impregnated cotton roll should be placed into the gingival sulcus for added protection, c. The cuff rubber dam technique should be employed to prevent slippage of the acid impregnated cotton pellets into the oral cavity, d. Very small cotton pellets or mini applicators must be used and excess solution should be dampened on gauze, e. Tweezers used to carry the cotton pellets must not be used for other purposes during the procedure.\nA reduction of the microhardness in both dentin and enamel has been reported following the use of trichloroacetic acid.[] Hence, restorations that will reinforce the weakened tooth structure are recommended. Chemical adhesion to tooth structure favours glass ionomer cement as the restorative material of choice.[] Recently a ‘reverse sandwich restoration’ comprising of microfilled resin composite and resin modified glass ionomer cement has been proposed to overcome the hydrolytic instability of glass ionomer cement. The microfilled resin tends to flex with the tooth thus reducing the chances of debonding.[]\nTrichloroacetic acid etches dentin and enamel and hence, conditioning is not recommended prior to insertion of the glass ionomer cement.[] However, refreshing the tooth surface with a bur is necessary because dentin that has been treated with trichloroacetic acid is severely demineralised and is not suitable for bonding with either dentin-bonding agents or glass ionomer materials.[] |
A 63-year-old African American man was transferred to the neurological intensive care unit from a nearby hospital after presenting with an acute history of multiple falls. Earlier that day, he had fallen once while walking to the bathroom from his bed and had fallen again shortly thereafter while walking through his living room. The patient's family decided at that time to bring him to the nearby hospital for treatment. At that hospital, no focal findings were discovered, but his blood pressure was noted to be elevated at 272/139 mm Hg. At that point, the original hospital placed the patient on a nicardipine drip to treat his hypertension and transferred the patient to our medical center, where he arrived late that night.\nThe patient presented to the medical center complaining of dizziness and agonizing headache. He was also noted to be somnolent and experiencing some dysarthria. The family at the bedside reported that the patient's past medical history was significant for hypertension, diabetes mellitus type II, and hyperlipidemia. It was also determined that the patient was a former chronic smoker, did not consume alcohol, and used marijuana occasionally. Physical exam showed some lethargy, dysarthria, right gaze paresis and nystagmus, vertical gaze weakness, and dysmetria on the right side more so than on the left. There was no hemiparesis, and visual fields were normal to confrontation. The patient had 5/5 strength, intact sensation, and 2+ reflexes of the upper and lower extremities bilaterally. It was determined that the patient had 4 points on both the NIH Stroke Scale and the Modified Rankin Scale for Neurologic Disability at admission. Computed tomography (CT) of the head and neck revealed decreased attenuation throughout most of the cerebellar hemispheres suggestive of ischemic injury (Fig ), diminished caliber of the fourth ventricle indicating increased pressure, and an old watershed infarct on the right side between the territories of the middle cerebral artery and the posterior cerebral artery (PCA). The area of the brain that is perfused by the PCA was surprisingly completely spared from infarct. CT angiography of the head revealed diffuse abnormally decreased attenuation of the cerebellar hemispheres with sparing of the vermis, suggesting infarction of both SCAs, and a narrow irregular left vertebral artery with a risk for subtotal thrombosis. There was no evidence of stenosis or a blood clot in the basilar artery or either SCA at the time of the CT angiography. Furthermore, it was discovered that the patient had a partial fetal-type PCA (fPCA) originating from the internal carotid artery (ICA) on the right side as seen in Figure .\nDue to the fact that the patient was well out of the window for thrombolytic therapy, he was initially treated with aspirin. Neurosurgery was consulted because of the patient's declining mental status caused by a mass effect in the posterior fossa. They elected to immediately take the patient in for a decompressive craniotomy. During the procedure, the dura overlying the cerebellum was noted to be very taut and to have a blue discoloration. Upon incision of the dura, there was spontaneous protrusion of necrotic brain matter. Much of the left and right cerebellum hemispheres were necrotic, but the vermis appeared to be spared from damage. The necrotic tissue was cleared, and the patient's cranium was closed.\nOver the next 24 h after the operation, the patient was extubated and remained lethargic but arousable. He responded to verbal commands, and facial movements were symmetrical. His right lateral gaze weakness persisted, and movement was diminished somewhat in the left extremities. Additionally, CT of the head showed new hydrocephalus, and electrocardiogram monitoring revealed intermittent atrial fibrillation throughout the day. Transthoracic echocardiogram was performed after discovery of the patient's arrhythmia, and it showed an ejection fraction of 55–60% as well as no evidence of residual thrombus. An amiodarone drip was administered in an effort to normalize the patient's heart rhythm. Anticoagulation with warfarin was not prescribed for another 3 weeks, as the patient experienced some bleeding in the posterior fossa after surgery. Neurosurgery placed an external ventricular drain to help decrease the intracranial pressure.\nPrior to the discovery of this patient's atrial fibrillation, the intensive care team had proposed that the cause of the stroke might have been an embolism from the diseased left vertebral artery. However, knowing that this patient was experiencing intermittent atrial fibrillation led the team to assess that the cause was more likely an embolism from the left atrium. Because it is unlikely that 2 separate emboli occluded both SCAs simultaneously, it was postulated that this was a single top of the basilar artery embolism that was large enough to occlude both SCAs. Such an occlusion should have led to ischemic injury of the regions of the brain perfused by the PCAs. Given this, the team determined that the bilateral PCA sparing may have been due to collateral perfusion supplied by the fetal origin of the PCA on the right side.\nThe patient remained lethargic over the next 24 h despite the placement of the external ventricular drain. The next morning, however, the patient woke up with markedly improved symptomatology. He was oriented to person and place, followed commands appropriately, and had 5/5 strength in all extremities along with intact sensation. Dysarthria and dysmetria of both upper extremities were noted, but overall the patient made a remarkable recovery from his condition the day before. The team continued to monitor and treat the patient over the next 3 weeks, and the patient was discharged for rehabilitation. At discharge, it was determined that the patient once again had 4 points on both the NIH Stroke Scale and the Modified Rankin Scale for Neurologic Disability. |
A 72-year-old male presented to emergency department with tender mass on lower back and posterior aspect of neck. Apparently, he had noticed it on his lower back 6 months ago and had grown in size with another similar swelling appearing on his neck, also they were increasingly painful and sore to touch. He had a history of ongoing sore throat for 3 years for which he was extensively investigated by ENT services. His CT neck and mediastinum did not show any abnormalities at that time. This was followed by laryngoscopy which showed a white patch on the right vocal cord, biopsy of which showed non-specific chronic inflammatory changes and cultures grew candida. Background history included COPD; he was heavy current smoker and smoked about 60 cigarettes per day. His medications included salmeterol/fluticasone propionate and ipratropium bromide inhalers. Vitals were within normal range. On examination, 4 cm nodular, firm, tender, immobile mass on lower back and 3 cm mass on posterior aspect of lower cervical spine were noted.\nHis laboratory investigation including renal and liver function, full blood count and coagulation profile were normal. A CT of thorax, abdomen and pelvis was arranged which showed 2 × 2 cm mass in right lung lower lobe likely malignant with no hilar or mediastinal lymphadenopathy, also 3 × 3 cm soft tissue mass at C6-C7 level posteriorly and 4 × 2 cm mass was noted on lumbar region with multiple prominent inguinal lymph nodes ().\nA referral for bronchoscopy was sent and biopsy of the nodule was planned. While awaiting bronchoscopy appointment, biopsy was taken from mass in lower back and send for histopathology. Histology showed poorly differentiated tumour cells in cohesive groups and sheets with moderate pleomorphism and eosinophilic cytoplasm; immunohistochemistry was positive for AE1/3, cytokeratin 7 and carcinoembryonic antigen antibody stain and periodic-acid schiff. Weak nuclear positivity for TTF-1 was also seen. Overall features were consistent with poorly differentiated adenocarcinoma likely of lung origin.\nHe was then referred to oncology services and unfortunately passed away within 5 months of diagnosis. |
A 34-year-old man presented to his family practitioner with a two-month history of swelling and discomfort in the left lateral lower leg just above his ankle. There was no preceding history of trauma. Physical examination revealed soft tissue fullness at the junction of the proximal two-thirds and distal one-third of the left fibula which was painful to touch. The patient was sent for an ultrasound for evaluation and possibly to aspirate a presumed ganglion cyst.\nUltrasound was performed using a General Electric Healthcare Logiq E9 linear ML 6–15 MHz transducer (GE Healthcare Wauwatosa, WI). A cortically based lesion was noted arising from the anterolateral cortex of the fibula with elevation of the periosteum and a thin rim of echogenicity surrounding the mass, presumed to be a thin shell of bone () which appeared intact without adjacent soft tissue mass. Two fluid-fluid levels were noted within the mass which were mobile on patient rotation indicating the cystic nature of the lesion's contents. No internal soft tissue mass extending from the fibular medullary canal was noted. There was increased vascularity in the adjacent soft tissues on color Doppler evaluation consistent with inflammatory changes. The most likely diagnosis was a cortically based aneurysmal bone cyst (ABC) and not a soft tissue solid or cystic mass. In view of the periosteal elevation or sonographic “Codman's triangle,” the mass was thought to be centered and to have originated within the bone rather than to be a soft tissue mass such as a ganglion cyst which had eroded into the bone. A Brodie abscess or subacute osteomyelitis could have this appearance as symptoms can be indolent, but these tend to be metaphyseal and centrally located. The adjacent fibular cortex was normal with no adjacent soft tissue mass to suggest an underlying aggressive bone lesion such as osteosarcoma or metastasis with secondary ABC formation.\nMRI was obtained for preoperative planning, consisting of axial and sagittal T1-weighted and fat saturated T2-weighted images with axial and sagittal fat saturated spoiled gradient echo sequence after gadolinium administration. This confirmed a cortically based mass arising from the fibula extending into the anterior compartment, with fluid-fluid levels seen on the T2-weighted sequence (). There was peripheral enhancement and increased T2 signal in the adjacent soft tissues and fibular marrow consistent with inflammatory changes corresponding to the increased vascularity seen sonographically. No primary lesion could be seen and this was presumed to be a primary cortical ABC.\nRadiograph demonstrated a cortically based lesion with a narrow zone of transition, elevating the periosteum with a thin shell of bone (). No other lesions were seen in the fibula or tibia. The patient underwent surgical excision of the lesion with curettage. A histologic diagnosis of aneurysmal bone cyst was made forming a 3.2 × 1.7 × 0.9 cm cystic mass (). Microscopically, a cystic space with peripheral shell of reactive bone with numerous osteoclast giant cells and scattered histiocytes and lymphocytes was noted. Some of the histiocytes contain hemosiderin. |
The patient involved provided written consent for reporting of this case.
A 63-year-old woman with medical history of super morbid obesity (BMI 54) and atrial fibrillation for which she was anticoagulated with apixaban presented for an open reduction internal fixation (ORIF) of an ankle fracture. Significant medical history included diabetes mellitus type 2, obstructive sleep apnea, chronic obstructive pulmonary disease, and diastolic heart failure. The patient's last dose of apixaban was 48 hours prior to surgery. Other than moderate anemia (hemoglobin 8.8 g/dL), all laboratory studies, including a coagulation profile, were normal.
Prior to surgery, the patient was offered a sciatic nerve catheter and an adductor canal block as part of a multimodal postoperative analgesia strategy. Because of her many, serious medical conditions, we concluded that a peripheral nerve block offered the best opportunity to provide satisfactory postoperative analgesia. Specifically, we were concerned that the postoperative pain management primarily with opioid medications would pose increased cardiopulmonary risk to the patient. We were careful to explain the risks associated with peripheral nerve blocks, including the risk of bleeding and hematoma formation, and verbal consent was obtained.
The surgery was performed under general anesthesia and her intraoperative course was uncomplicated. Upon arrival to the recovery room, our acute pain service was contacted to evaluate her for peripheral nerve blockade. We positioned the patient in the lateral decubitus position and placed a sciatic nerve catheter. Though technically challenging due to body habitus, this sciatic nerve block was performed successfully and without any complication. The patient was then positioned supine for the adductor canal block. The leg was externally rotated and the knee slightly flexed for optimal positioning. A high-frequency linear array ultrasound transducer was applied to the mid-thigh in short-axis and the adductor canal was identified. Imaging was again challenging given the patient's habitus, but with firm compression of the ultrasound transducer, the important anatomical structures were clearly identified. The superficial femoral artery (SFA) was visualized |
A 32 year old female patient, mother of two children, poorly built and nourished reported to our clinics for replacement of her missing teeth. Patient also complained of burning sensation in the mouth since 1 yr. Patient had visited our department 2 years back with the complaint of pain in the gums and mobile teeth. Diagnosis of acute necrotising ulcerative periodontitis was given. Teeth were not in a condition to be saved and all her teeth were extracted on follow up visits. Patient also gave the medical history of amenorrhea after the delivery of her 2nd child 6 years back.\nClinical examination showed severely resorbed maxillary and mandibular alveolar ridges. Erythema of hard palate, maxillary alveolar mucosa and distobuccal portion of mandibular alveolar mucosa bilaterally was present with foci of ulcerations (Fig. ). On palpation mucosa over the hard palate was non tender, soft and oedematous in consistency with no discharge from the lesions. Noma was given as provisional diagnosis and tuberculous ulcer, ulcerative stomatitis secondary to malnutrition, chronic major apthous ulcers and necrotising sialometaplasia were considered as differential diagnosis. Occlusal and panoramic radiograph, routine blood investigations, test to rule out tuberculosis and HIV and incisional biopsy were carried out. Panoramic radiograph revealed extensive bony destruction in the maxilla and mandible. Blood investigations showed increased erythrocyte sedimentation rate and decreased haemoglobin level. White cell count and random blood glucose value were within normal range. Mantoux test was negative and HIV antibody tests were non-reactive.\nHistopathologic study revealed diffuse dense chronic inflammatory cell infiltrate and cells with reniform nuclei suggestive of abnormal Langerhans cells in the connective tissue. To confirm the lesional Langerhans cells, immunohistochemical staining for CD1a which is diagnostic for it was performed () (Fig. ). The result was positive and final diagnosis of Langerhans cell histiocytosis was given. To evaluate the generalized bony involvement plain radiography of skull, chest and limbs were done and no abnormality was detected. Bone scan was carried out further and the results revealed increased tracer uptake in the mandible, pelvis: left iliac bone and right pubis, bilateral femori, right proximal femur, bilateral tibiae and abnormal uptake in the lumbar vertebrae (Fig. ). Endocrine opinion was taken and investigations revealed altered levels of thyroid stimulating hormone, prolactin, follicle stimulating hormone and luteinizing hormone (). Patient was diagnosed to have prima-ry hypothyroidism and hyperprolactinemia. Opinion of oncology department was taken and ultrasonography of abdomen was advised. Ultrasonography revealed bulky pancreas with hypoechoic echotexture. Based on histological and other previously mentioned positive investigatory findings the diagnosis of LCH with systemic involvement was established.\nPatient was referred to the department of medical oncology. Chemotherapy was planned, including 6 cycles of vinblastine (10 mg weekly) and systemic prednisolone (10 mg thrice a day) for 6 weeks. For oral lesions no treatment was undertaken except for 0.12% chlorhexidine daily rinse. Hormonal replacement therapy was planned after the chemotherapy.\nBefore third cycle of chemotherapy patient’s total leucocyte count (TLC) fell down to 730/cu mm and granulo-cyte- colony stimulating factor( 300 mcg subcutaneously) was given following which TLC raised to 65,990/cu mm. Third cycle was administered but the patient’s condition deteriorated, patient was brought to the emergency unit in a semiconsciousness state. Her random blood glucose level, blood urea nitrogen, serum creatinine was high and electrolytic imbalance was present. Unfortunately patient succumbed to the complications of the disease. |
A 68-year-old male with known stage IV sarcomatoid renal cell carcinoma presented to the emergency department (ED) complaining of shortness of breath; his vital signs were unremarkable with stable oxygen saturation, and his physical examination had been normal. Because of the high suspicion of pulmonary embolism related to his risk factors, a CT scan of the chest with IV contrast was done, which had shown bilateral segmental and sub-segmental PE and a cardiac mass in the right ventricle that is consistent with known renal cell carcinoma as shown in Figure . There was no right ventricular (RV) strain on CT or transthoracic echo, and troponin and B-type natriuretic peptide (BNP) had been normal. Given that he was stable, a decision was made to start him on therapeutic anticoagulation with low molecular weight heparin with close follow-up.\nSix months later, he presented to the ED with worsening shortness of breath. A CT scan was repeated at that time, showing an increased clot burden in the pulmonary arteries at segmental and sub-segmental levels but now with evidence of new RV strain on CT despite him being on a therapeutic dose of anticoagulation. It was thought that this was not related to hypercoagulability, but instead, these were tumor emboli related to his cardiac metastasis.\nCardiothoracic surgery was consulted, and as recommended, cardiac MRI was done to see if the cardiac metastasis could be removed surgically. Cardiac MRI showed a large right ventricular mass attached to the endocardium on the RV septal aspect and the anterior aspect of the RV outflow tract (RVOT); the mass was highly mobile and prolapsing into the pulmonary artery in real-time view (Figures -).\nSurgical option was considered because the mass was near the RVOT and was prolapsing into the pulmonary arteries. It was feared that massive tumor embolization could happen later on, leading to a high-risk pulmonary embolism and cardiac arrest. The patient also had good functional status despite having stage IV renal cell carcinoma.\nUnfortunately, before surgical planning, he was admitted with fatal intra-abdominal bleeding from the tumor, as shown in Figure , and passed away despite angiographic embolization and resuscitation. |
A 26-year-old pregnant women of Asian Indian origin was referred to the obstetric casualty by the general practitioner with a provisional diagnosis of placental abruption in view of abdominal pain, pallor and inability to find fetal heart. The women had insidious onset of pain for 12 hrs, and with reduced fetal movements for the same duration. The woman was third gravida with 32 weeks gestation, with previous two normal deliveries and an uneventful antenatal period. Her dating and anomaly scan were normal. There was also no history of trauma, any diagnostic or therapeutic intrauterine intervention, vaginal bleeding or labor pains. She had no significant past medical, surgical history or gynecological history.\nOn examination, she was pale with respiratory rate of 28 per minute, pulse rate of 110 per minute and blood pressure of 90/60 mmHg. On abdominal examination, uterus measured 30 weeks, with mild abdominal tenderness and no increased tone. The uterine wall felt thin but with regular outline and no fetal part was palpable. Fetal heart could not be localized. Speculum examination showed no vaginal bleeding and there was marked tenderness on internal examination and cervix was uneffaced and os admitted tip of finger.\nUltrasound scan revealed fetal demise. An intact amniotic sac with fetus and normal volume of amniotic fluid, without any echogenecity to suggest blood, was seen lying outside the uterus. Uterus was deviated towards left iliac fossa. Diagnosis of rupture uterus with fetal demise was made and the woman was prepared for laparotomy. Blood investigations revealed hemoglobin of 7.7 gm% with no evidence of coagulopathy.\nAfter resuscitation with intravenous fluids, the patient was taken for surgery. Emergency exploratory laparotomy was done with consent for hysterectomy or uterine repair with tubal sterilization under intravenous antibiotic cover. An intact amniotic sac with fetus was found along with 1.5 liter of haemoperitoneum. Membranes were ruptured and male stillborn fetus (weight 2.2 kilograms) was delivered with breech extraction. The amniotic fluid was clear, and placenta was lying outside the uterus and appeared complete and normal. There was a 3 inch tear on the right side of the uterine fundus, 2 cm anterior to the cornua. The myometrium adjacent to this was relatively thin. There was no evidence of couvelier uterus. The tear was repaired by suturing the uterus in double layer. Bilateral tubal ligation was done. She received one unit of blood during intra-operative and three during the postoperative period. Her postoperative period was uneventful and was discharged after eight days. |
Our patient is an African American 55-year-old male who presented to Saint Louis University Hospital (SLUH) in November 2014 with lower back tightness and aching right-sided lower back pain radiating to the right flank for the prior two weeks. Initial ultrasound and CT imaging was positive for a right-sided mixed solid and cystic retroperitoneal mass with severe mass effect on adjacent organs. The mass, which replaced the right kidney, was noted to be 18.5 cm × 17.8 cm × 20.5 cm, and there was presence of retroperitoneal lymphadenopathy (Figures and ). CT imaging of the chest was negative for pulmonary mass or adenopathy. A recommendation for a full body PET-CT and MRI of the head was made by urology and oncology to assist with the staging of the malignancy, but neither was completed due to patient intolerance of the studies.\nAt the time of initial presentation, our patient was an inmate at the local prison. For this reason, he requested finishing his sentence before beginning management of his condition. After returning to SLUH one month later, repeat CT imaging evidenced a new 6.2 cm × 4.2 cm nodular soft tissue mass in the left extraperitoneal space which appeared to invade the urinary bladder. There was also evidence of a new 1.7 cm liver lesion and 7 mm nodular thickening along the right posterolateral bladder wall. A PET-CT with sedation confirmed a large renal mass with intense fludeoxyglucose (FDG) uptake and was positive for mediastinal and bilateral hilar, external iliac, and inguinal lymphadenopathy, a right lower lobe pulmonary nodule, lesions in the right and left hepatic lobes, and diffuse osseous metastatic disease. There was intense uptake in the bladder appearing contiguous with the prostate and penis (Figures , , and ). This FDG uptake within the prostate was initially concerning for a synchronous primary prostate carcinoma; however a prostate exam revealed a small and smooth prostate that was inconsistent with malignancy. Multiple magnetic resonance images demonstrated no evidence of intracranial metastatic disease throughout this time. An image guided biopsy of the renal mass and immunohistochemistry evidenced spindled, malignant cells (Figures and ) with cytoplasm positive for vimentin, CD10, and AE1/AE3 (Figures , , and ) and negative for EMA, CD31, RCC, and desmin which supported a diagnosis of sarcomatoid renal cell carcinoma (SRCC). There was no histologic evidence within the biopsied material to determine a specific subtype of RCC. For this reason, our patient was given a diagnosis of unclassified renal cell carcinoma with sarcomatoid features.\nA review of systems for our patient after this second presentation was notable for new onset distal leg edema, constipation, and severe penile pain with dysuria. He denied urinary retention or priapism. Physical exam demonstrated a hard mass at the base of the penis without ulcerations or rash. Due to the aggressive nature of SRCC and the extent of metastasis, the patient was not a candidate for cytoreductive surgery including penectomy. He was started on chemotherapy with gemcitabine and doxorubicin with pegfilgrastim. He was discharged to a skilled nursing facility for continued chemotherapy with a prognosis of less than one year. |
A 62-year-old woman (Figure , II-3) was admitted to the hospital for recurrent partial headache with weakness of one side and aphasia for about 45 years. In her first attack, the patient suddenly experienced an aura with visual disturbances which she described as increasing scotomata in the bilateral visual field. After a few seconds, the patient developed a serious headache, mainly located on the left side. After a few minutes, she presented a paralysis of the right side and speech difficulties accompanied by dizziness and vomiting. These symptoms resolved after about 2 h. After this initial onset, she had an attack nearly every 4–5 years, and the clinical presentations of her attacks were always similar to the first one. The duration of the aura symptoms and the migraine was typically 1–2 h but sometimes the migraine could last up to 4 days. Sometimes headaches occurred before the hemiplegia and aphasia. Each headache was accompanied by dizziness and vomiting but without loss of consciousness. In most attacks, this patient experienced additionally a flushing of the neck and face and felt that the skin temperature of this affected area was increased, but the temperature was never measured. These symptoms may be related to an extracranial vasodilation when a migraine attack occurred. She did not undergo regular treatment except for simple analgesics as a symptomatic therapy. Recently, her condition aggravated as the frequency of attacks increased from once every 4–5 years to once every 1–2 weeks which had a serious impact on her everyday life. Therefore, during a severe migraine attack, she visited our hospital. We reviewed her family history, and we found that three other subjects, her mother, brother, and nephew, had similar clinical symptoms (Figure ). Their presentations are as follows:\nThe proband's mother (Figure , I-2) died of uremia at the age of 72. According to her husband and children, she reported typical hemiplegic migraines since an age of 14 years with five attacks per year on average. The aura symptoms were similar to those in the proband, including bilateral visual symptoms (scotomata), speech difficulties, and hemiparesis.\nThe proband's 55-year-old brother (Figure , II-1) had first at the age of 15 headache attacks with nausea, vomiting, visual field defects, and one-sided motor weakness. Usually, these attacks last 5 h. The disease presentation was progressive with age.\nThe proband's nephew (Figure , III-1), a 25-year-old fitness coach, had first headache attacks with visual symptoms (scotomata) and lateralized motor weakness at the age of 13. Each attack lasts about 20 min.\nAfter admission, her neurological examinations were unremarkable and brain magnetic resonance imaging (MRI) and Magnetic Resonance Angiography (MRA) showed no meaningful abnormalities (Figure ). Thus, the suspected diagnosis was transient ischemic attack (TIA). During her hospitalization, the patient had several migraine attacks that were characterized first by visual symptoms, then aphasia and right limb paralysis 10 min later, and finally severe headaches after 20 min. At that time, the neurological examination revealed: no loss of consciousness, motor aphasia, muscle strength 2 in the right limb, and normal findings in the examination of the residual nervous system. After about 1 h, the symptoms of the aura were relieved, while the headache lasted for about 1 day. However, the symptoms were not relieved after dual antiplatelet aggregation treatment, and transthoracic echocardiography and carotid ultrasound failed to identify any underlying cerebrovascular etiology. After careful consideration of all aspects, she was diagnosed with hemiplegic migraine. So, we conducted a genetic test on the patient and found a heterozygous point mutation (c.4495T>C) in exon 26 of the SCN1A gene. This mutation caused amino acid 1499 to change from phenylalanine to leucine (p. Phe1499Leu), which may cause the disease by affecting the SCN1A protein function.\nTo establish the diagnosis, we performed a genetic test on those family members to analyze for the presence of mutations in genes including CACNA1A, ATP1A2, and SCN1A related to FHM. We only found a gene mutation in SCN1A, but this mutation was detected in all affected subjects in this family (Figure ). Therefore, this patient was diagnosed with FHM3. She was discharged after receiving a health education on migraine attacks, which suggested her staying away from stress, bright lights, sleep disturbances, physical exertion, and alcohol consumption because these have all been reported as trigger factors in FHM (). Upon being discharged from the hospital, she had intermittently taken flunarizine capsules and rizatriptan benzoate tablets to prevent and control migraine attacks. After 6 months of follow-up, the efficacy of the drug was uncertain, because the frequency of headache attacks was not adequately reduced. After the low efficacy of her medication became clear, we consulted again the literature and consider now a trial with lamotrigine or acetazolamide (). |
A 30-year-old female originally from Slovenia was first noted to have asymptomatic hypertension in 2008 during a routine check-up at a gym. She was referred initially to a general physician who performed a 24-h blood pressure monitor, which revealed a mean systolic pressure of 160 mmHg and a mean diastolic pressure of 110 mmHg. A work up for a secondary cause of hypertension included a renal ultrasound, which demonstrated a normal renal and urinary collecting system, a normal aldosterone:renin ratio and an echocardiogram which showed evidence of moderate left concentric hypertrophy. A 24-h urine collection for caetacholamines and metanephrines was carried out and found to be markedly elevated (see ).\nThe patient was then referred to our service for further evaluation. The patient had no past medical history and was not on regular medication. She had been living in Ireland for the past 7 years and was attending a course at a local college. There was no family history of hypertension or endocrine disease. On examination, she had no clinical features to suggest an endocrinopathy and, apart from an elevated blood pressure of 150/100, her clinical examination was entirely normal. A full biochemical profile and endocrine blood panel including neuroendocrine markers were carried out (see ). Radiological localisation with a CT adrenal protocol was carried out and showed a left sided 4.5×3.2 cm adrenal mass with Hounsfield units of 35. A MIBG scan confirmed increased and abnormal uptake by the left-sided adrenal lesion with no other uptake noted. The patient underwent a laparoscopic adrenalectomy after adequate alpha and beta blockade. The patient was re-evaluated 6 weeks after the surgery. She remained normotensive of all medication and repeated urinary caetacholamine and metanephrine collections were within the normal range. Genetic analysis was performed and confirmed a mutation in exon 8 of the RET gene.\nExamination of the thyroid gland revealed no clinical abnormality and a pentagastrin stimulation test was carried out. This demonstrated an elevated basal calcitonin and a peak calcitonin of a 156 ng/l post 0.5 μg/kg of pentagastrin by injection (see ). A thyroid ultrasound showed a normal-sized thyroid gland with no radiological abnormalities. The patient underwent a total thyroidectomy and histology, which confirmed the presence of C cell hyperplasia but no focus of medullary thyroid carcinoma (see ).\nTo date, there is no biochemical evidence of primary hyperparathyroidism. We have been unable to provide genetic counselling to first-degree relatives as they are not living in Ireland, but correspondence has been sent to the family regarding the genetic mutation identified. At present genetic analysis is not available to the family, but the mother of the index case has undergone a total thyroidectomy with histology showing evidence of a 0.8×0.8×0.6 cm focus of medullary thyroid carcinoma in the left lobe of the thyroid, with no evidence of extra-thyroidal extension. |
A 17-year-old white female gravida 1 para 0-0-0-0 with a past medical history of depression, panic attacks and right nephrolithiasis presented to a rural community hospital at 38 weeks 4 days for induction of labor. She had been receiving routine prenatal care. Patient’s pregnancy was complicated by right nephrolithiasis, young maternal age, a slipped disc in the lumbar spine and chlamydia during pregnancy. The decision was made to proceed with induction of labor at 38 weeks and 4 days rather than waiting for 39 weeks secondary to the patient’s nephrolithiasis and back pain from a slipped disc. This decision was made with the assistance of physicians who were board certified in maternal fetal medicine. Medications at the time of induction included sertraline 100 mg daily, macrobid 100 mg daily for urinary tract infections, and prenatal vitamins. She claimed compliance with these medications and denied any other medications, vitamins, or supplements. She had previously been prescribed escitalopram for her depression and anxiety but switched to sertraline 50 mg daily 4 months prior to delivery. Prior to this she had been on escitalopram for greater than two years. She felt that symptoms were not adequately controlled, so the dose of sertraline was later increased to 100 mg daily 2 months and 17 days prior to delivery. The last 100 mg dose of sertraline was given 5 h and 26 min prior to delivery. The patient denied use of tobacco, alcohol, or illicit drugs during pregnancy.\nPatient was induced via 10 mg topical vaginal dinoprostone given one time and allowed to dilate. Subsequently she underwent an amniotomy after she was placed on oxytocin 20 unit in 1000 mL sodium chloride 0.9% infusion given at the rate of 1munit/min. She was allowed to progress through the normal stages of labor with no maternal or fetal complications. Continuous fetal monitoring was performed per hospital protocol, and tracing fluctuated between category I and category II without any unexpected decelerations or tracing abnormalities. At no time was cesarean section considered as fetal status appeared reassuring. There was no indication for biophysical profile during labor. Delivery occurred spontaneously. After delivery of the head it was noted that there was a loose nuchal cord which was easily slipped over the head of the infant and resolved. She gave birth vaginally to a 3373 g male infant at 0226 with APGAR scores of 3 at 1 min, 6 at 5 min and 8 at 10 min.\nAt the time of delivery, the newborn exhibited motor depression, cyanosis, and minimal respiratory effort. It was noted that the umbilical cord only had two vessels upon inspection. A pulse oximeter was placed at 2 min to monitor oxygenation and at four minutes it was noted that the infant was euglycemic with a blood sugar of 110 mg/dL. At nine minutes the infant continued to exhibit grunting, retractions, and tachypnea consistent with respiratory distress. He was found to have a respiratory rate of 30 and 82% oxygen saturation. He was then placed on 10 L of supplemental oxygen via simple bag mask. At ten minutes the APGAR score was 8, with points taken off due to cyanotic extremities and continued poor respiratory effort. The infant was then transferred to the neonatal intensive care unit (NICU) at 0240 for further evaluation and monitoring. In the NICU there was a sustained period of hypoglycemia which was initially noted at 0605 with a blood glucose of 42 mg/dL. Other than this, laboratory values and physical exam was normal for the duration of the hospitalization. The neonate remained on 2 L oxygen via nasal cannula until 1200 and was lowered to 1 L oxygen via nasal cannula which was continued for an additional 3.5 h. At this point the infant was weaned off supplemental oxygen and would not require it for the remainder of the hospitalization. The hypoglycemia persisted due to poor feeding with measurements of 63 mg/dL at 1202 and 68 mg/dL at 1803. At this point both the oxygen saturation and blood sugars both normalized and would continue to stay within normal limits until the patient discharged the following day at 1430. The patient was instructed to follow up with the pediatrician 2 days after discharge.\nThe pediatrician noted that there were no symptoms of respiratory distress or complications of the hypoxia in the newborn at 4 days postpartum. |
A 56-year-old woman, a mother of 3 children, presented to the surgical department with a history of multiple neck lumps of 4 months duration. She also had generalised vague abdominal pain, loss of appetite and lower back pain. She had no significant medical, family or psychosocial history. Clinical examination revealed multiple, bilateral enlarged cervical lymph nodes which were firm to hard in consistency. Thyroid examination revealed a 2 × 2 cm firm lump on the lower pole of the left thyroid lobe. Examination of other lymph node groups revealed enlarged right inguinal lymph nodes. Abdominal examination and rectal examination were normal. Vaginal examination revealed a hard, unhealthy uterine cervix. Breast and axillary examination were unremarkable. She had spinal tenderness, but the neurological examination of the lower limbs was normal.\nHer basic blood investigations, liver profile and renal functions were within the normal limits. Ultrasound scan of the abdomen revealed no abnormalities. Ultrasound scan of the neck revealed multiple nodules in the thyroid, with increased vascularity, and multiple enlarged cervical lymph nodes with obliterated fatty hila suggestive of malignant deposits. Contrast enhanced computed tomography of the neck, chest, abdomen and pelvis showed a mass in the uterine cervix (Fig. ) with multiple enlarged lymph nodes in the inguinal, iliac, para-aortic, anterior mediastinal and bilateral deep cervical groups (Fig. ). There were multiple low-density nodules in the thyroid gland (Fig. ). A mixed density mass lesion was also noted in the lower pole of the left thyroid lobe (Fig. ). Furthermore, an anterior wedge fracture of the L2 vertebra was seen, probably secondary to bone metastases.\nBiopsies from the uterine cervix and endometrial curettage revealed moderately differentiated squamous cell carcinoma, signifying local extension of the cervical carcinoma into the endometrium (Fig. ). Excision biopsy of a left cervical lymph node revealed metastatic deposits of moderately differentiated squamous cell carcinoma similar to that of the uterine cervix (Fig. ). Ultrasound-guided fine needle aspiration cytology of intra-thyroid nodules revealed malignant squamous cells (Fig. ). This was followed by a panendoscopy of the upper aerodigestive tract which yielded negative results.\nA multidisciplinary meeting with the oncologists, surgeon, gynaecologists, pathologists and radiologists was conducted. Considering the histopathological and imaging findings with negative panendoscopy, a diagnosis of squamous cell carcinoma of the uterine cervix metastasising to the cervical lymph nodes and thyroid gland was made. A decision was made to commence palliative chemoradiotherapy. After 6 months, the patient developed generalised body oedema. She was transferred to a specialised cancer treatment centre for further palliative management, where she passed away a short while later, due to acute renal failure secondary to ureteric obstruction. |
An 18-year-old male with history of Rothmund-Thomson syndrome diagnosed at the age of two presented with acute right mid leg pain while cutting to kick the ball during a soccer game. He reported a history of prior right leg fracture four years ago sustained while playfully wrestling with his younger brother. After the prior fracture healed, the patient noticed a bump at the level of his mid right shin, which corresponded to the site of the patient's current leg pain. He denied any pain in the right leg prior to sustaining the fracture, and he denied any other preexisting medical conditions.\nPresenting radiographs of the right tibia-fibula showed mild deformity of the right tibia from prior healed fracture with mildly displaced and comminuted fracture through the mid diaphysis of the tibia (). Focal anterior cortical thickening of the tibia was present at the level of the acute fracture. The fracture was treated with closed reduction and casting. Serial follow-up radiographs obtained over the next 13 months showed delayed and incomplete union across the tibia fracture (). At the 13-month follow-up, additional radiographs of the left tibia were obtained to evaluate a focal painless bump of the mid left shin the patient reported developing shortly after his right tibia fracture. Left tibia radiographs showed focal anterior cortical thickening of the mid tibia diaphysis corresponding to the palpable bump (). No fracture line or lytic bone lesion was appreciated. The patient did not return for additional follow-up.\nThe patient returned to clinic 4 years later for treatment of a left olecranon fracture sustained during a fall playing soccer 3.5 weeks earlier. Presenting radiographs showed a mildly displaced subacute fracture through the left olecranon (). There was early cortication along the fracture margins and more osteolysis than expected along the fracture line given the reported age of the fracture. Therefore, a bone biopsy of the fracture was performed, which was negative for neoplasm and infection. Tension band wiring of the olecranon fracture was subsequently performed (). Early remodeling without bridging bone formation was present on two-month follow-up radiographs (). The patient did not return for any additional follow-up of his elbow fracture.\nThe patient again returned to clinic approximately 5 years following the olecranon fracture with acute right mid leg pain after landing on the right leg awkwardly while jumping during a soccer game. Radiographs showed a mildly displaced acute fracture through the area of prior fracture 9 years earlier (). An MRI with and without contrast of the right leg was performed to exclude underlying malignancy at the fracture (). No bone tumor was present on the MRI.\nRepeat radiographs of the left tibia-fibula were also obtained due to the patient reporting a gradual increase in size of the bump on his left shin he had first noticed eight years earlier. The radiographs showed increase in the focal cortical thickening at the anterior mid tibia with development of a stress fracture through the area of cortical thickening (). An old ununited transverse fracture through the left lateral malleolus was incidentally noted on the tibia-fibula images (). Upon interviewing the patient again, he reported injuring his left ankle 6 years earlier while stepping off a truck. He treated the ankle injury with compression dressing without seeking professional medical treatment. He could not recall any other trauma to the left ankle.\nThe patient was treated with closed reduction and casting of the right tibia fracture (), and he was discharged on crutches to remain on non-weight bearing on the right leg for 6 weeks. Since the patient's left tibia stress fracture was asymptomatic, no treatment recommendations for the stress fracture were made. |
A 20-year-old right-handed male had accidentally amputated both hands in a paper cutting machine []. The patient was declined by two major hospitals before reporting to us. The relatives were counselled about the complexity of the case, its outcome, and our limitations in a small nursing home set-up. Consent was taken accordingly both from the patient and relatives. Emphasis was given to replant the dominant right hand on priority.\nReplantation of the right hand was started first while the left hand was kept preserved in the cooling chamber of the fridge. Dissection of the amputated right hand was started in the sequence of skeletal fixation with axial K-wires, repair of ulnar artery in the Guyon's canal, radial artery, three dorsal veins, nerves and flexor tendons []. The second surgical team to assist the senior author joined the procedure and carried out debridement, skeletal fixation and vessel preparation. The ulnar artery, first dorsal metacarpal artery and 3 dorsal veins were repaired on the left hand. Because of severe blood loss during and after amputation, the patient was haemodynamically unstable and was resuscitated with Ringers lactate and plasma expanders. Due to haemodynamic instability and unavailability of blood during surgery, the repair of the tendons and nerves on the left hand was deferred. The patient was stabilised postoperatively with 3 units of blood. The entire procedure was done under bilateral brachial block, which lasted for 14 h.\nThe patient had some amount of tissue necrosis from the left thenar region which took about a month to heal, and the patient was lost to follow-up. He however reported back after two months. There was minimal flexion in the right hand and all the joints in both the hands had become stiff. After physiotherapy for 1 month, the patient underwent secondary surgery for the unrepaired tendons and nerves.\nBy the end of 6 months, the patient was able to perform his activities of daily living. After 15 months, he had good sensory recovery. He was able to wear clothes, perform toilet activities, write [Figures –], lift heavy weights, ride bicycle and harvest paddy in the field and perform the activities of daily living [].\nThe functional outcome was assessed by using the Chen's criteria which includes - Grade I (excellent), Grade II (good), Grade III (fair) and Grade IV (poor).\nThe details of sensory and motor recovery in our case are as follows:\nAccording to Chen's criteria, the patient had excellent function in the right hand and good function in the left hand []. |
A 25 year old G2P1001 sub-Saharan African married seamstress at 8 weeks amenorrhea, presented to a primary level hospital in Cameroon with 4 days history of lower abdominal pains and vaginal bleeding. She had no known chronic illness and denied having any past history of pelvic inflammatory disease or assisted reproduction.\nShe reported being well till 4 days prior to presentation when she started experiencing abdominal pain; the pain was mainly in her lower abdomen, dull in nature, non-radiating, mild in intensity and was initially intermittent then became constant. It was associated with mild per vagina loss of bright red blood. She had no other symptoms. This prompted her to consult at a drug store, where she was prescribed phloroglucinol tablets 80 mg/day in 2 divided doses which she took for 3 days but had no regression of the symptoms. The persistent lower abdominal pain and mild vagina bleeding prompted a second consultation at our health facility. Upon presentation she was haemodynamically stable and vaginal examination was relevant for a closed cervix. A presumptive diagnosis of threatened abortion with possible aetiology of a urinary tract infection was made. Urinalysis showed leucocyturia while obstetric echography revealed an empty gestational sac measuring 29.9 mm. Diagnoses of blighted ovum and urinary tract infection were made. She was given a short course of antibiotics and programmed for manual vacuum aspiration. Vacuum aspiration was done and the patient served a single intramuscular dose of 10 units of oxytocin. The bleeding and lower abdominal pain stopped and she was discharged the next day.\nTwo weeks following discharge lower abdominal pain reoccurred. This time, the abdominal pain was constant and localized to the left lower quadrant. It was associated with intermittent episodes of bright red vaginal bleeding. These symptoms persisted for 3 days and prompted another consultation.\nOn arrival she had a good general state. Her blood pressure was 102/64 mmHg, heart rate 88 beats/min, respiratory rate of 20 breaths/min, temperature 37.2 °C, O2 saturation at 97% and weight 58 kg. Her conjunctivae were pink and no scleral icterus, heart sounds were normal and lung fields clear. Her abdomen was flat, moved with respiration and there were no scars. There was no tenderness on superficial palpation but left iliac fossa tenderness on deep palpation. The liver and spleen were not palpable. A speculum examination was unremarkable but for bright red blood oozing out of the cervical os. Vaginal examination revealed a firm closed posterior cervix with left adnexal tenderness on bimanual palpation. The limbs were without particularity.\nBased on these findings a tentative diagnosis of an associated ectopic pregnancy was made. A repeat pelvic ultrasound revealed: a homogenous uterus; left adnexal mass measuring 58 mls; pouch of Douglass collection. A repeat positive pregnancy test and sonographic findings confirmed a diagnosis of ectopic pregnancy.\nThe patient was immediately planned for an emergency exploratory laparotomy indicated for a possible ectopic pregnancy. Preoperative work up included: normal haemoglobin of 11.7 g/dl; normal white cell count of 8100/µl; normal platelet count of 320,000/µl, normal kidney function test (serum creatinine of 0.64 mg/dl and urea of 12.7 mg/dl), glycaemia of 85.9 mg/dl and normal serum electrolytes of: (sodium 134 mmol/l, potassium of 4.17 mmol/l and chloride of 103 mmol/l).\nAn emergency laparotomy was performed under general anaesthesia. Intra-operative findings revealed unruptured left tubal pregnancy (Fig. ) in the ampulla. A left salpingectomy was performed with excision of the unruptured fetus. The right tube was inspected and found to be normal. The abdomen was then closed layer by layer. The patient was monitored in the recovery room for 1 h during which she was haemodynamically stable. She was sent to the ward where close monitoring continued.\nPostoperatively, the patient was maintained on nil per os, intravenous infusions of dextrose–saline, intravenous prophylactic antibiotics and analgaesics. Early ambulation was encouraged upon recovery from general anaesthesia. Routine postoperative care in the ward continued and was uneventful. She was discharged on post operation day 7. Evolution thereafter was favourable. |
This 82-year-old woman with myasthenia gravis was seen at a local hospital complaining of dyspnea. Two days later her dyspnea worsened (PaO2 76.3 mmHg, PaCO2 99.1 mmHg). She underwent noninvasive positive-pressure ventilation; however, it was unable to correct dyspnea. Therefore, she subsequently underwent tracheal intubation and placement of a central venous catheter in the right internal jugular vein. Although her blood gas analysis improved (PaO2 143.0 mmHg, PaCO2 32.7 mmHg) she remained unable to breathe on her own. The development of a myasthenic crisis was suspected and she was admitted to our hospital the next day. Steroid pulse therapy and an immunoadsorbent technique were performed. Her respiratory condition was stable on the 9th hospital day and she was extubated. On the 10th hospital day the attending physician who had come to exchange the central venous catheter noticed a decline in her consciousness. She had been seen to brush her teeth 30 min earlier but the onset of the consciousness decline was uncertain. An emergent brain CT scan (), obtained 30 min after her consciousness decline was noted, revealed air inflow in the cortical veins, the bilateral cerebral hemispheres, the bilateral cavernous sinuses, and the sella turcica, consistent with the anterior intercavernous venous sinus. A chest CT scan showed air in the left brachial vein. CAE was diagnosed and she was treated with oxygen inhalation and intravenous injections of edaravone. Hyperbaric oxygen therapy could not be delivered because she showed a deterioration of consciousness and was too unstable for transfer to a facility with a hyperbaric chamber. The air density was markedly diminished on a brain CT scan obtained 50 min later () and not observed on brain CT scans acquired 18 hr after the first scan (). MRI scans were performed a week later. DWI showed multiple areas of restricted diffusion affecting predominantly the cortical areas in the bilateral hemispheres adjacent to air, the corpus callosum, and the cerebellum (). A transesophageal echocardiogram (TEE) with injection of agitated saline was normal. Her consciousness level improved gradually although she manifested paralysis of the left leg and both arms. She was transferred to a rehabilitation hospital 3 months later. |
A 75-year-old man with a past medical history of diabetes mellitus was admitted to the Emergency Department of our University Hospital. He had a history of acute low back pain in the region of the lumbar spine in the last 4 days before his admission to the hospital. Two days before his admission he experienced lower leg weakness and fever (oral temperature 38.5°C). Clinical examination showed neck stiffness. After initial evaluation and brain CT scan – which revealed no damage – he had a lumbar puncture. The patient hospitalized with the diagnosis of meningitis (CSF: 765 white cells per cubic millimeter, elevated protein level: 70 mg per deciliter, decreased CSF glucose levels: 35% of serum glucose). Staph. aureus was cultured from cerebrospinal fluid (CSF) sample.\nThe neurologic condition of the patient impaired very quickly and at the end of the third day, after his admission, he developed paraplegia. Deep tendon reflexes were absent in the lower limbs and severely diminished in the upper limbs. After neurosurgical consultation an emergency magnetic resonance imaging scan (MRI) of the brain and the whole spinal spine was performed, five days after the admission of the patient to the hospital. It revealed a contrast-enhancing subdural mass collection posterior and left lateral to the spinal cord at the level L2 – L4 which was compressing the spinal cord. It also revealed arachnoiditis in the whole thoracic and lumbar vertebral body of the spinal cord. After intravenous contrast administration there was an intense enhancement on the boundaries of the collection and widespread meningeal enhancement (figures and ). Brain MRI with intravenous contrast revealed no intracranial abnormalities.\nMeanwhile, at the end of the fifth day, the condition of the patient impaired with respiratory failure and quadriplegia and he was admitted to the ICU. The patient remained alert and cooperative. Laboratory data showed a leukocytosis of 20,000/mm3 with a left shift, median elevated serum alkaline phosphatase (789 IU/l) and decreased albumin (2.8 g/dl). Also the C-reactive protein was elevated (17.5 mg/dl).\nA L2–L4 laminectomy with midline incision of dura and arachnoid was performed eight days after the admission of the patient into the hospital. The purulent material of the abscess was observed posterior and left lateral to the spinal cord and unfortunately extended in the whole lumbar vertebral body of the spinal cord (according to the surgeon, there was possibly an empyema to the whole vertebral body of the spinal cord). An empyema was extended to lumbar nerve roots and to the psoas muscles. The purulent material was removed at the levels of laminectomy and the vertebral body copiously irrigated superiorly and inferiorly with saline solution. The wound was closed, and a usual drainage system was placed (inflow/outflow drain). Cultures from the purulent material and the blood were positive for staph. aureus.\nDespite the removal of the purulent material and the appropriate antibiotic treatment (IV vancomycin, meropenem, fluconazole) the neurologic condition of the patient declined immediately after the operation and he developed severe impairment of consciousness. Except respiratory failure, which was always a problem, hemodynamic instability was also reported during his ICU stay. In ICU, all failure systems were supported. The patient was well hydrated, he was fed with enteral nutrition and he had an early tracheostomy in an attempt of weaning from mechanical ventilation. Inotropic and vasoactive agents were needed to stabilize mean arterial pressure >65 mmHg. The patient died 6 weeks after his ICU admission. |
A 17-month-old first-born female child presented to the brachial plexus clinic with complaints of weakness of the right shoulder and elbow, noticed from birth. Hand and wrist movements were present at birth. No improvement in the movements over these 17 months was reported. The paresis was accompanied by a prominent scar over the entire limb from the shoulder to the thumb. Both the thumb and index finger were also hypoplastic, apart from the scarring []. The skin scar was reported to be bright red at birth (no pictures were available) that had lightened over time. The child also had a ptosis of the right eye with lighter pigmentation of the iris as compared to the other eye. The iris was reported to be paler at birth and had apparently improved in pigmentation with growth. The reported developmental milestones were appropriate for her age.\nThe mother had developed chickenpox after the 3rd month of pregnancy (week 13) that had been treated symptomatically. No antivirals or immunisation had been administered. The family history was non-contributory.\nOn examination, the general development was confirmed appropriate for age. The following clinical features were noted:\nThe eye: Ptosis, miosis and enophthalmos were present in the right eye []. The iris was light brown in colour compared to black on the left side The skin: The skin over the entire pre-axial border of the upper limb was scarred from the shoulder to the hand, including the hypoplastic thumb and index fingers Right upper limb: The limb was held in the classic ‘waiter's tip’ position described for an upper brachial plexus palsy. No active shoulder abduction or external rotation was present. Adduction and internal rotation were present but weak. The elbow was held fully extended with no active flexion and had good extension power. The wrist and the fingers showed full movements. The motor profile, as per the Active Movement Scale, is summarised in . Of note was the complete absence of co-contractions. Given the age, a reliable sensory examination could not be carried out.\nWith a provisional diagnosis of CVS with brachial plexus palsy, we performed a magnetic resonance scan to rule out cervical root agenesis or cervical cord anomalies. It confirmed no anomaly in the plexal elements and revealed severe atrophy of the biceps and shoulder musculature.\nGiven the age of the child, an exploration of the brachial plexus was not indicated. However, an Oberlin's nerve transfer could possibly be performed to attempt restoration of elbow flexion. Hence, the arm was explored for the status of the musculocutaneous nerve. During surgery, the biceps was found to be completely pale and had undergone a severe fatty degeneration []. Hence, the plan of nerve transfer was abandoned. The thumb was made more functional by phalangisation []. The first web was released by a Gilles' hatchet flap, and a full-thickness graft was placed.\nTo restore elbow flexion, a provisional free functioning muscle transfer is planned after the age of 4 years. Tendon transfer options for the shoulder, like a Trapezius transfer, need to be assessed as the child grows up. Similarly, the cosmetic correction of the skin scar and the ptosis is planned for later. |
A 41-year-old woman presented with a mass on the left upper lip and difficulty in pronunciation. The mass developed after she bit the upper lip 5 years earlier. The volume of mass was not reduced; however, the patient complained of pain. One year after the development, she visited an otolaryngologist. The mass was diagnosed as mucocele and aspirated. However, only blood was aspirated from this lesion, and the lesion's size was not reduced. Two years after aspiration, the size of mass increased, and she visited a plastic surgeon. The lesion was diagnosed as hemangioma by magnetic resonance imaging (MRI). Ethanol was injected into the lesion twice. Although the lesion was slightly reduced at the first injection, it did not change at the second. She consulted a dental clinic two years after the second injection and then was referred to our department.\nOral examination revealed a circumscribed submucosal single nodule, approximately 30 × 20 mm in size in the left upper lip. The overlying mucosa was smooth, with bluish discoloration. On palpation, the nodule was elastic firm and mobile (). Cervical lymph nodes were not palpable. MRI revealed a relatively well-demarcated lesion in the left upper lip. The lesion showed low signal intensity on T1-weighted images; however, it had a high signal area suspecting the subacute bleeding image in the centre of tumor. The lesion showed mostly high signal intensity on T2-weighted images (). Under the clinical diagnosis of hemangioma, surgical enucleation was performed under local anesthesia. The tumor was removed with ligation and ablation of the inflow blood vessels. The overlying mucosa was partly removed, and the wound was closed by sutures (). Postoperative course was uneventful. The patient was free of recurrence 2 years after surgery ().\nThe size of excised the lesion was 30 × 20 mm. The specimen had reddish brown surfaces covered by thin-walled capsule including the mucosa of partial lower lip and inflow blood vessels which were well demarcated. Microscopically, the lesion was a well-circumscribed mass surrounded by fibrous connective tissue and showed a variety of cellularity imparting a lobular architecture in low power (Figures –). The lesion was characterized by irregular cavernous spaces and solid cellular areas. The cavernous spaces contained erythrocytes and were lined by a single layer of flattened endothelial cells. Large cavernous spaces were filled with a mix of erythrocytes and organizing thrombi. The solid areas showed proliferation of spindle cells arranged haphazardly or in short interlacing fascicles. Epithelioid cells were also seen, some of which contained large cytoplasmic vacuole.\nImmunohistochemically, most endothelial cells lining the cavernous spaces, spindle cells within solid areas, and epithelioid cells within both areas strongly reacted with vimentin (Figures and ). The endothelial cells lining the cavernous spaces reacted strongly with CD34 (), CD 31, factor VIII, smooth muscle actin (SMA) (), and Wilms tumor-1 (WT-1) (). The spindle cells within solid areas focally reacted with CD34 (), CD31, SMA (), and WT-1 (), whereas epithelioid cells were positive for SMA (), WT-1 () and negative for CD34 (), CD31. S100 protein, AE1/AE3, D2-40, and EMA were negative in endothelial cells, epithelioid cells, and spindle cells. From these findings, the lesion was diagnosed as SCH. |
A 65-year-old female presented with a 6-week history of vomiting. This had progressed to complete intolerance of all solid and liquid oral intake, except for small sips of water. The patient also described epigastric discomfort followed by a band-like pain around the circumference of her thorax, at the level of the epigastrium. Her LAGB had been performed at another institution 19 years prior. There was subsequent tightening of the gastric band three years following initial placement. Due to geographical relocation of the patient, there was no formal follow-up. The patient reported acceptable weight loss and maintenance, with no adverse symptoms. She had an unremarkable abdominal examination. Computed tomography imaging identified oral contrast pooled above the level of the laparoscopic band, at a much lower level in the stomach than expected. The small and large bowel were collapsed and almost gasless, suggesting band slippage and absolute obstruction (Fig. a and b). Exploratory laparoscopy and removal of the gastric band was performed on the following day.\nExtensive adhesiolysis was required intra-operatively to mobilize the left lobe of the liver from the stomach. The tube of the adjustable gastric band could be seen tracking into the stomach wall near the greater curvature, with the band completely internalized in the stomach. Dissection along the gastric tube into the lumen of the stomach allowed removal of the internalized gastric band with an endocatch bag. A 3-cm gastric defect on the anterior stomach wall was closed with laparoscopic sutures. Gastroscopy was then attempted during the same procedure. At the gastro-oesophageal junction, there was an obstructing lesion presumed to be scar tissue and fibrosis that had surrounded the internalized gastric band, however it was also biopsied to exclude malignancy. The obstruction could not be traversed with the endoscope. There was a presumed small patency as gastric insufflation was possible via the gastroscope. Given this gastric inlet obstruction, a decision was made to place a surgical gastrostomy tube for gastric decompression and to allow enteral nutrition.\nThe patient was discharged on the sixth day post operatively. Five weeks following the initial procedure, the patient underwent repeat gastroscopy. The obstructing lesion was not reduced in size and still could not be passed by the gastroscope. Histopathology of the biopsies taken was that of a low grade invasive adenocarcinoma. Staging investigations with computed tomography and positron emission tomography identified the distal oesophageal lesion extending in to the stomach, along with bilateral lung nodules likely inflammatory in nature however potentially early metastases. The patient received neoadjuvant chemotherapy and radiotherapy prior to operative resection. |
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