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A one month old baby girl was brought by her mother to the lactation clinic for further evaluation due to the difficulty of breastfeeding. The mother complained of pain in the nipple which was present throughout the entire breastfeeding session and usually recurred with each episode of breastfeeding. Two weeks after delivery, the mother noticed a tooth on the baby’s lower left gum. A week later, she noticed an ulcer under her baby’s tongue, which occasionally bled.\nIn light of the continuous pain during breastfeeding, the mother became reluctant to continue and infant formula milk was given as a substitute. Self-examination by the mother did not reveal any nipple crack or breast injury. Her antenatal history was uneventful. There was strong family history of natal teeth, and this baby’s siblings had similar problems.\nOn examination, the mother’s breast appeared to be normal and consistent with a lactating breast. There was no inflammation or engorgement. Some mild eczema was noted around the nipple, but there was no infection. Cervical and axillary lymph nodes were not palpable.\nExamination of the oral cavity of the baby revealed a neonatal tooth over the left anterior region of the mandibular ridge. The tooth measured 2 mm × 1 mm in size, was whitish opaque, and had Grade II mobility (Fig. ). There was also a whitish ulcer over the ventral aspect of the tongue measuring 1 mm × 1 mm (Fig. ). The parents declined radiological investigations and tongue biopsy for the baby. Hence, the clinical diagnosis was neonatal tooth associated with Riga-Fede disease causing difficulty in breastfeeding.\nThe baby was then referred to the pediatric dental surgery department. After a thorough discussion with the parents, the neonatal tooth was extracted under local anesthesia. The tooth had a crown but lacked a root. Following extraction, the baby did not have any complication such as bleeding and infection. The wound healed well within 2 days and she successfully resumed taking breast feeds.\nCurrently, at 10 months post-extraction, the child is growing well and still breastfeeding. Examination of the oral cavity revealed that only one central lower incisor tooth was present (Fig. ). |
A 61-year-old Caucasian female presented to her general practitioner with painless and palpable right-sided submandibular and thyroid swellings. Her past history revealed left mastectomy and axillary clearance with postoperative chemoradiotherapy for grade 2 lobular breast carcinoma 2 years ago, and left radical nephrectomy for clear cell type RCC (Fuhrman grade 2, stage pT1, completely excised with no renal vein involvement) with no regional or distant metastases 7 years ago. Considering her recent history of invasive breast carcinoma, she was referred onto the breast surgical clinic for further assessment. An ultrasound scan (US) of her neck suggested multinodular thyroid goitre (largest nodule in right lobe) and a well-defined hypoechoic mass in the right submandibular gland. US-guided fine-needle aspiration (FNA) cytology of the right thyroid nodule was nondiagnostic on two separate occasions and of the right submandibular gland suggested a pleomorphic adenoma (PA).\nShe was then seen in our otolaryngology department where a complete ear, nose, and throat examination was normal except for the obvious right submandibular and thyroid gland swellings. A computed tomography (CT) scan of her neck was performed which revealed a tumour mass in the right submandibular gland with prominent vascularisation unusual for benign lesions such as a PA (). CT scan also showed a large nodule in the right thyroid with similar intense vascular enhancement as the submandibular lesion (). There was no suspicious cervical lymphadenopathy. A bone scan showed no osseous metastases and chest X-ray revealed no pulmonary metastases.\nInitially the right submandibular gland was excised revealing an encapsulated tumour within the glandular tissue. A right level I and II neck dissection was also performed with preservation of the marginal mandibular nerve. Histopathological examination of the tumour showed striking similarity to the clear cell RCC of left kidney resected previously (). The diagnosis was further supported by immunohistochemical findings that showed positive staining for vimentin, CD10, low molecular weight cytokeratins, epithelial membrane, and RCC antigens, and negative for carcinoembryonic antigen (CEA), S100, and CD34. Subsequently the patient's case was discussed at the combined surgical oncology and pathology meeting, and due to the suspicious nature of the thyroid gland, a total thyroidectomy was performed with preservation of the recurrent laryngeal nerves and parathyroid glands. Histopathological analysis of right lobe of thyroid gland confirmed metastatic clear cell RCC similar to the submandibular gland disease (). As there was no extracapsular spread and no lymphatic disease, no further adjuvant therapy was given to the patient. At 6-month postoperative followup, the patient showed no signs of recurrence or other metastases. |
A 74-year-old woman presented with severe dyspnea (NYHA 4). She had had an anterior wall myocardial infarction several years ago and at that time, she was diagnosed as having three-vessel disease with left ventricular aneurysm. Subsequently, an implantable cardioverter defibrillator (ICD) was inserted due to ventricular tachycardia. She also had chronic kidney disease and diabetes mellitus and her left ventricular ejection fraction was 28%. Chest X-ray showed increased vascular markings suspicious of pulmonary edema. Chest computed tomography was performed and pulmonary edema was diagnosed (). Mitral flow showed a relaxation abnormality pattern with a prominent L wave, and estimated right ventricular systolic pressure was 51 mmHg. Interestingly, echocardiographic examination 5 months previously when she was admitted to the hospital due to milder symptom of dyspnea (NYHA 2) showed a restrictive physiology () and more advanced diastolic dysfunction. Despite efforts to reduce pulmonary edema by administering diuretics, the patient's symptom did not improve and in fact became further aggravated. Chest X-ray showed an increase in pulmonary vascular markings and consolidation despite intensive diuretic therapy (). Subsequently, a sputum culture revealed Klebsiella pneumoniae. After intensive antibiotic therapy rather than diuretics, the patient's symptoms improved.\nDyspnea is defined as difficult or labored breathing and occurs whenever the work of breathing is excessive. It is a common symptom in a variety of cardiac and noncardiac diseases, emphasizing the need for a thorough differential diagnosis. A comprehensive history, physical examination, and basic laboratory tests are important in the initial assessment; however, the diagnosis may depend on specialized testing. Cardiac causes of dyspnea include right, left, or biventricular congestive heart failure with systolic dysfunction, coronary artery disease, recent or remote myocardial infarction, hypertension with left ventricular hypertrophy, cardiomyopathy, valvular dysfunction, pericardial diseases, arrhythmia, congenital heart disease, and primary diastolic dysfunction. In an elderly population with high rates of comorbidities, determining whether symptoms are attributable to cardiac conditions or to other coexisting comorbidities can be challenging. In this case, despite the presence of significant diastolic dysfunction with elevated filling pressure, the severity of the patient's dyspnea and the degree of diastolic dysfunction and filling pressure elevation did not correlate well when compared with serial echocardiographic examination. Thus, a discordant relationship between echocardiographic parameters of diastolic dysfunction and filling pressure elevation and the severity of the symptom of dyspnea can provide insight into the cause of the dyspnea in the presence of multiple comorbidities. |
A 24-year-old woman (gravida 2, para 1) who was 34 weeks + 5 days pregnant was admitted to the emergency department of our hospital on 24 July 2018 because of acute upper abdominal pain. The pain had begun the previous day after she had eaten her dinner, and was associated with nausea and vomiting. She had no history of fever. She had had a normal-term delivery 6 years previously. She had undergone bilateral laparoscopic distal salpingectomy because of bilateral oviduct and ovarian cysts 2 years previously. Her menstrual cycle was regular. She had undergone IVF-ET in December 2017 because of tubal-factor infertility caused by bilateral salpingectomy. Two fresh embryos had been transferred. Transvaginal ultrasonography had revealed a dichorionic and diamniotic twin pregnancy with normal cardiac activity and gestational sac situated in a normal uterine cavity. The subsequent course of prenatal care was uneventful. She had no other medical, surgical, or gynecological history.\nOn admission to our hospital, the patient was in a semi-recumbent position and could not lay down. Her pulse rate was 120 to 140 beats per minute. Her blood pressure varied from 110/70 to 120/80 mm Hg, and her respiratory rate varied from 20 to 25 breaths per minute. Her body temperature was normal. She did not appear pale or feel faint. An abdominal examination revealed mild tenderness in the upper abdomen and a firm abdomen. We did not detect uterine contractions or increased uterine tone. The fetal heart rate was >160 beats per minute. A vaginal examination showed that the cervix was closed, and the fetal membrane was intact. There was no evidence of bleeding or abnormal discharge.\nEmergent ultrasonography showed two live fetuses, one in a cephalic presentation and the other in a scapular presentation. The parameters of fetal growth corresponded to 34-week gestation. There were no placental abnormalities. On Doppler examination, vascular flow and amniotic fluid volume were normal. Fetal heart rate ranged from 162 to 178 beats per minute. Laboratory blood tests revealed the following: white blood cells, 16.9 × 109/L; neutrophils, 91.5%; red blood cells, 3.79 × 1012/L; hemoglobin, 127.00 g/L; platelets, 272.0 × 109/L; and creatinine, 79 μmol/L. Tests for urine and liver function yielded normal results. There was no evidence of coagulopathy. Abdominal ultrasonography showed fluid (depth, 5.6 cm) surrounding the appendix, and low-dose abdominal computed tomography (CT) revealed a small amount of abdominal effusion. Low-dose chest CT examination showed a small amount of pericardial effusion. As the pain had persisted despite antibiotic and other symptomatic treatments administered by another doctor in the emergency department on the day before admission, a fetal non-stress test (NST) was performed. The NST showed that fetal heart rate in the right fetus was persistently >160 beats per minute and that normal baseline variation of the heartbeat had seriously declined (<5 bpm; Fig. ). Similar changes were seen in the left fetus (Fig. ).\nA diagnosis of pregnancy complicated by acute abdomen was considered, and emergent exploratory laparotomy and cesarean section were performed. During surgery, hemoperitoneum (3000 mL) was found in the upper abdominal cavity. After exploration of the intestinal loops, a rupture was palpated on the right side of the uterine fundus, which was covered with blood clots. A lower-segment cesarean section was performed. The fluid of both amniotic sacs was mildly polluted by meconium. Two live infants were uneventfully delivered from the cephalic presentation. The Apgar scores of the older, male infant were 4, 7, and 7 at 1, 5, and 10 minutes. His weight was 2800 grams. The Apgar scores of the female infant were 4, 7, and 8 at 1, 5, and 10 minutes; her weight was 2120 grams. The uterine fundus and cornua were symmetrical, and both placentas were distal to the rupture site. The intact placenta was delivered spontaneously. Uterine examination performed after blood clot removal showed that the 5-cm–wide rupture site was located next to the right cornual area, 7 cm from the proximal end of the right fallopian tube (Fig. ). The distal ends of the fallopian tubes were absent. The proximal ends of the fallopian tubes, the ovaries, and the left cornu were normal. The rupture site was repaired with two layers of continuous vicryl sutures and 1 layer of continuous vicryl 2–0 sutures. Total intraoperative blood loss was approximately 400 mL. Two units of packed red blood cells, 400 mL fresh frozen plasma, and 6 units of cryoprecipitate were transfused during the operation. There were no further complications.\nThe postoperative course of the mother was uneventful, and she was discharged on the seventh postoperative day in good condition. The infants required cardiopulmonary resuscitation, intubation, and mechanical ventilation, after which, their vital signs promptly recovered. Both infants were transferred to a tertiary care center and survived. The female infant was discharged from the neonatal intensive care unit on the 14th day of life, without further complications. The male infant was diagnosed with left hydronephrosis and required an operation. He was discharged from the neonatal intensive care unit on the 30th day of life and transferred to the neonatal unit for urinary surgery. Follow-up assessments of the mother and the female baby on the 42nd postpartum day yielded normal results, and an ultrasound examination of the maternal uterus showed normal puerperal changes after cesarean section. The patient had no further complications, but she was advised to avoid future pregnancies because of the high risk of recurrence. |
A 25-year-old housewife, who occasionally worked on a fishing boat, sustained injuries after an accident. As she was lowering the fishing net, the rope of the fishing net rigidly wound around her arm. Her arm remained twisted at the bow of the boat for approximately 10 minutes before help came and she was released. The patient was immediately transported to a nearby hospital and hospitalized.\nAt the time of hospitalization, the patient reported of severe pain on her right elbow. We found that the pain had severely restricted movement. We observed edema on the patient’s elbow and forearm. There was overall tenderness, but distal sensory, motor, and circulation were normal. The patient did not have a previous history of trauma pertaining to either the elbow or forearm. She had not experienced any pain or discomfort of the elbow or forearm before the accident.\nRadiographs revealed anterior dislocation of the radial head and avulsion fractures at the medial epicondyle and at the humeral tip. tip. We observed 6° of volar angulation of the ulna, crossing from the metaphysis to the ulnar shaft, in contrast to 6° of PUDA observed in the unaffected arm. We did not observe indicators of chronic radial head dislocation, such as radial head deformity or degeneration (). Incidents of plastic deformation of the ulna in adults are very rare. To exclude neglected dislocations and to assess soft tissue injury around the joint, we conducted magnetic resonance imaging (MRI). We found that the proximal radioulnar joint and the radial head showed neither dysplastic or degenerative changes. Torn annular ligament parts were found floating in hematoma, and fractures, probably induced by the avulsion at the flexor tendon insertion, were observed at the medial epicondyle and the humeral tip (). In spite of the radiographic suggestions against chronic but for acute plastic deformation of the ulna, we concluded that it may be too rash to perform surgical treatment. So we performed manipulation via compression of the radial head from the anterior aspect, which had already been attempted at the emergency unit but had failed, to restore the radial head and to test the plausibility of conservative treatment. As before, we were able to restore the dislocation relatively easily when the forearm was in a neutral position, but even a slight extension of the elbow or supination of the forearm led to re-dislocation with a popping sound. Subsequently, we decided that the patient required surgical treatment.\nMonteggia fractures in adults generally require surgical treatment. Acute Monteggia fractures are treated through open reduction of the ulnar shaft, as the first line of treatment, to achieve anatomical reconstruction. Open reduction alone generally leads to satisfactory reduction of the radial head. But when radial head reduction, in spite of surgical treatment, is unsatisfactory or when there is soft tissue involvement, such as the annular ligament or the nerves, open reduction of the radial head through the lateral approach or annular ligament reconstruction is recommneded []. In addition, the effectiveness of corrective osteotomy for restoring PUDA have already been established for the treatment of ulnar deformity or of nonunion-induced radial head dislocations []. Thus, taking together preexisting guidelines and the literature on the treatment of fractures in pediatric patients, we decided to measure the magnitude of PUDA of the unaffected arm and use it as an anatomical marker of recovery after ulnar osteotomy. We aimed to achieve complete radial head reduction through ulnar osteotomy, but in the case that the reduction is unstable we planned to use the lateral approach for the reduction and perform adjunct annular ligament reconstruction.\nWe made another attempt to reduce the radial head dislocation through posterior compression. As our attempt before, although the dislocation was easily restored, even a slight supination of the forearm or extension of the elbow led to redislocation with a popping sound.\nWe made a 15-cm incision at the posterior aspect of the forearm to expose the ulnar shaft. We then located and marked the apex dorsal angulation with a C-arm. Using a saw, we osteotomized the region encompassing the posterior cortical bone to the apex of the deformity. To prevent unnecessary rotational deformity and changes in length, we intentionally preserved a portion of the anterior cortical bone. Aiming for a slight overcorrective of PUDA, we fixed the plate (3.5 mm locking compression plate, 7 holes; DePuy Synthes, West Chester, PA, USA) at a 10° tilt around the central screw hole, so that the tip of the tilt was on top of the site of osteotomy. And we inserted one cortical screw each into the proximal and distal screw hole nearest to the site of osteotomy. We tightened the screws in alternation so that the proximal and distal ulnar fragments came in contact with the plate and ensured that the remaining anterior cortical bone was osteotomized (). Post-corrective ulnar osteotomy, we found that slight compression of the radial head led to reduction of the radial head. We inserted locking screws into the remaining holes to complete the fixation. Using C-arm imaging, we checked that manipulation in any motion (rotation of the forearm, extension or flexion of the elbow, or any motion under forearm rotation) did not redislocate the radial head. This meant that adjunct treatment such as annular ligament reconstruction or resection of medial or lateral soft tissue was not required. Postoperative radiographs revealed that the degree of PUDA of the unaffected and affected sides were 6° and 7°, respectively.\nDuring the first postoperative week, the patient was administered cast immobilization at 90° of elbow flexion. The patient then carried out joint motions under hinged brace administration for 5 weeks. On the 3rd postoperative month, the patient’s elbow and forearm range of motions (ROMs) were fully restored with respect to the unaffected side. We observed the following ROMs: 0° of extension, 145° of flexion, 80° of pronation, and 70° of supination. Radiographs also revealed bone union of the ulna (, ). At the final 3-year follow-up, we surgically removed hardware from the patient who at the time was asymptomatic and did not show any signs of degenerative changes. |
A 46-year-old man also reported exercise/activity-related pain and fatigue from early childhood. As patient 1, he was always considered to be “a lazy child”. He was not able to run and physical activities such as walking or swimming were challenging. As a child he reported trying to build a good relationship with his physical education teachers, so they would feel pity for him and he could avoid any strenuous physical activities (PA). He experienced three severe episodes of rhabdomyolyses in his life. The first one happened during childhood, which followed vigorous physical activities. The second rhabdomyolysis episode was at the age of 18 following multiple squats, which he performed during a physical test for military service. Not qualifying for the military service, instead he had to complete a civilian service at the university hospital at the age of 22. As he was clearly weaker than his colleagues, a diagnostic investigation took place, which included a skeletal muscle biopsy. At that time, doctors advised him to avoid excessive physical effort. He was told about the second wind phenomenon; however, it was not explained how to reach it. Additionally, he was recommended to eat a maximum of 20% of his daily food intake in carbohydrates. However, the rest of the macronutrients recommendations were not specified. In the following years, his physical activity level decreased, leading to physical deconditioning, loss of muscle mass, and increase in fatty adipose tissue and body weight.\nThe third rhabdomyolysis episode was experienced one year ago and was nearly fatal. He was undergoing physical assessment for the insurance company and pushed himself too hard, resulting in a severe contracture of his lower back muscles. He was admitted to hospital. During the hospital stay he was also diagnosed with myocarditis. Following this episode, he decided to learn more about the condition to manage it better.\nHe began doing gentle aerobic exercises, which improved his ability to attain a second wind. He attended a conference where he met a Spanish team from whom he learnt about the strength training trial performed in Madrid and decided to try it himself in a local gym []. Initially he approached a personal trainer. However, he did not feel the trainer understood his condition and was pushing him too hard. He decided to write his own programme based on strength training principles and exercises he learnt at the conferences and meetings.\nWhen in the gym he reports always doing an aerobic “warm up” by cycling on a stationary bike for 20 minutes, he then exercises on resistance machines (chest press, seated row, butterfly, chest horizontal adduction, reversed butterfly, rare deltoids and trapezius, lat pulldown, leg press, leg adductors, and leg abductors). He finishes his sessions with more aerobic exercise by walking on a treadmill and/or cycling on a stationary bike. and illustrate his progress during the first three months of training (based on personal records). He completes between 5 and 15 repetitions of each exercise with one-minute rest in between sets. He stops a set of exercise earlier if he feels any discomfort in the muscle.\nHe did not report Delayed Onset Muscle Soreness (DOMS) or McArdle symptoms following initial sessions. After three months of resistance training, he found his sleep pattern, overall stamina and McArdle symptoms improved. He is now able to walk two kilometres stopping only once, as opposed to previously when he would have to stop multiple times. He has also found it easier to perform everyday tasks such as changing a car tyre. He has been eating a balanced diet. He autonomously chose not to take any supplements containing glucose pre- or intratraining session. |
A 32-year-old man with a remarkable history of polysubstance abuse presented to the emergency room with a 2-week history of diplopia. Six weeks earlier, the patient had presented to the emergency department after being found unconscious secondary to a suspected drug overdose and a question of possible physical assault and head injury. Toxicology panel was positive for cocaine, cannabinoids, and opioids. The patient was observed and improved, returning to his neurological baseline level after overnight monitoring. He was unable to recall additional details of the event and was discharged the day after his presentation. Cranial imaging during this initial encounter was not obtained by the trauma team. Approximately 4 weeks after his initial presentation, the patient reported to his primary care physician because he was experiencing diplopia. Brain MRI was obtained as part of an outpatient workup 2 weeks following symptom onset, and the sagittal T1 sequence revealed an abnormal rim – hyperintense core – heterointense lesion on the sagittal T1 sequence []. Fluid-attenuated inversion recovery (FLAIR) T2 sequence more specifically revealed a 6 cm × 3.6 cm × 3 cm hyperintense [], diffusion restricting, and apparent diffusion coefficient hypointense extra-axial collection without surrounding edema [ and ]. Significant mass effect on the temporal lobe and left midbrain [] was observed, and the patient was referred to the emergency department for further evaluation.\nAt the time of neurosurgical evaluation, the patient complained of double vision with downward gaze in the bilateral lower visual quadrants. He denied any alleviating or exacerbating factors and described it as constant throughout the day. The patient was otherwise healthy, with no other medical conditions. Contrast-enhanced CT of the head was obtained to assess for the progression or change in the lesion observed on MRI and demonstrated the similar contrast rim-enhancing lesion with significant surrounding mass effect [ and ]. Given the history of possible remote assault as well as aforementioned imaging findings, the lesion was suspected to be a chronic epidural hematoma. There was also consideration given that this might be an abscess due to the patient’s history of polysubstance abuse, but no clinical signs or symptoms of infection were noted.\nDue to the significant size and mass effect on the left midbrain in addition to the diplopia, the patient was taken for a left temporal craniotomy for evacuation and removal of the lesion. A small temporal craniotomy was performed. On removal of the bone flap, normal dura was not clearly visualized. Instead, a rather thick, yellow fibrous-like tissue was observed []. This tissue was carefully dissected, and specimens were sent for pathological analysis. Underneath this tissue, a consolidated subacute hematoma clot was found. The clot was removed and another layer of the thick, yellow tissue was encountered, which was also carefully dissected. Once this capsule of tissue was removed, normal dura was encountered. Interestingly, no inflammatory or purulent material was encountered during the procedure. No clear sources of bleeding, including arteries or veins, were identified. The cavity was copiously irrigated with antibiotic instilled irrigation. The dura was tacked up; the bone flap was placed back; and the wound was secured. Pathology results revealed an organized hematoma with marked fibrosis.\nPostoperatively, the patient remained neurologically unchanged and was observed in the intensive care unit for 2 days. On postoperative day 1, a CT scan of the head showed gradual parenchymal expansion into the void that had previously been occupied by the hematoma. The patient was discharged on postoperative day 5. At the 6-week postoperative follow-up visit, he had near-complete resolution of his diplopia and complete resolution of his epidural hematoma on head CT [ and ]. |
The first patient is a young woman, 20 years old at the time of diagnosis of a synovial sarcoma that originated from the soft tissue of the left hip. The neoplastic mass was surgically removed in May 2012, and the histological examination showed a synovial sarcoma with close surgical margins. Consequently, adjuvant chemotherapy with Doxorubicin and Ifosfamide was administered for a total of six cycles. During follow-up, in February 2015, the CT scan showed almost 15 nodules in the lung ranging from 5 to 10 mm (). In the same month the patient started therapy with Trabectedin at the dose of 1.5 mg/m2 every 3 weeks. The first radiological evaluation in April 2015 showed a dimensional reduction of all the lung nodules (). The patient continued the therapy, and in July and October 2015 the CT scan confirmed the partial response of the lung lesions.\nIn February 2016, after 16 cycles, the patient developed hematological toxicity with thrombocytopenia G2 and neutropenia G2. From the 17th cycle the dose was reduced to 1.2 mg/m2 (80% of the expected dose). In June 2016, after 20 cycles, the CT scan still showed stable disease. The patient asked for a therapeutic pause in order to complete her university studies and recover from the treatment's toxicity. Therefore, she started a follow-up program with a CT scan every 3 months.\nIn September 2017 (13 months after last dose) two lung nodules appeared to be increasing. After a multidisciplinary discussion with dedicated surgeons and radiotherapists it was decided to treat the two lung lesions with stereotactic radiotherapy. The treatment was well-tolerated. However, 6 months later, in March 2018 the radiologic findings showed disease progression with multiple, new lung metastases (). In April 2018, after a new multidisciplinary discussion, it was decided to restart systemic therapy with Trabectedin. The subsequent radiological evaluations in July and October 2018 showed stable disease with signs of response in some nodules (). The patient is continuing Trabectedin, without new significant adverse events. In the meantime, she successfully graduated and started her first work experience. |
A 43-year-old female had attempted suicide in the morning by burning charcoal and was found unconscious in the evening by her son. She had been diagnosed as having depression for 8 years while her son had mild intellectual disability. She was taken to the emergency room of our university hospital. It was estimated that it took about 27 hours from the time of the suicide attempt to the time of admission. The level of carboxyhemoglobin was within normal range at that time. HBOT (2.5 ATA for 120 min) started immediately after admission, and her unconsciousness gradually resolved and returned to the normal level. However, after the thirteenth session of HBO, it was interrupted to treat her wound because there was a second-degree burn mainly in her upper left limb. Her disorientation and abnormal behaviors appeared about twenty days later after interruption of HBOT. She could not open the door and try to eat hand cream. The EEG at that time showed predominant slow waves (3~7 Hz theta and delta) with poor alpha (8 Hz) waves, and MRI at that time showed diffuse hyperintensity of the bilateral hemispheric white matters consistent with changes of delayed leukoencephalopathy as a result of prior CO injury with hyperintense change of the bilateral globus pallidi (). Three sessions of HBOT were thus performed immediately. Eleven days later, she was transferred to our department for the purpose of detailed examinations and treatment of CO intoxication. Since HBOT and burn treatment could not be performed at the same time, the policy was to give priority to burn treatment. Thirty-two days later, burn treatment was finished and HBOT was thus restarted. During 136 sessions of HBOT, her neuropsychiatric symptoms gradually ameliorated. Her clinical improvement went side by side with the improvement in scores of FAB as observed in case 1. 326 days after her suicide attempt, she was discharged from our university hospital. At that time, the score of FAB was 18/18 and the EEG was normalized with dominant alpha (9 Hz) waves. At the present time, her neuropsychiatric conditions are almost well controlled for about two years and half after her suicide attempt. |
A 46-year-old man also reported exercise/activity-related pain and fatigue from early childhood. As patient 1, he was always considered to be “a lazy child”. He was not able to run and physical activities such as walking or swimming were challenging. As a child he reported trying to build a good relationship with his physical education teachers, so they would feel pity for him and he could avoid any strenuous physical activities (PA). He experienced three severe episodes of rhabdomyolyses in his life. The first one happened during childhood, which followed vigorous physical activities. The second rhabdomyolysis episode was at the age of 18 following multiple squats, which he performed during a physical test for military service. Not qualifying for the military service, instead he had to complete a civilian service at the university hospital at the age of 22. As he was clearly weaker than his colleagues, a diagnostic investigation took place, which included a skeletal muscle biopsy. At that time, doctors advised him to avoid excessive physical effort. He was told about the second wind phenomenon; however, it was not explained how to reach it. Additionally, he was recommended to eat a maximum of 20% of his daily food intake in carbohydrates. However, the rest of the macronutrients recommendations were not specified. In the following years, his physical activity level decreased, leading to physical deconditioning, loss of muscle mass, and increase in fatty adipose tissue and body weight.\nThe third rhabdomyolysis episode was experienced one year ago and was nearly fatal. He was undergoing physical assessment for the insurance company and pushed himself too hard, resulting in a severe contracture of his lower back muscles. He was admitted to hospital. During the hospital stay he was also diagnosed with myocarditis. Following this episode, he decided to learn more about the condition to manage it better.\nHe began doing gentle aerobic exercises, which improved his ability to attain a second wind. He attended a conference where he met a Spanish team from whom he learnt about the strength training trial performed in Madrid and decided to try it himself in a local gym []. Initially he approached a personal trainer. However, he did not feel the trainer understood his condition and was pushing him too hard. He decided to write his own programme based on strength training principles and exercises he learnt at the conferences and meetings.\nWhen in the gym he reports always doing an aerobic “warm up” by cycling on a stationary bike for 20 minutes, he then exercises on resistance machines (chest press, seated row, butterfly, chest horizontal adduction, reversed butterfly, rare deltoids and trapezius, lat pulldown, leg press, leg adductors, and leg abductors). He finishes his sessions with more aerobic exercise by walking on a treadmill and/or cycling on a stationary bike. and illustrate his progress during the first three months of training (based on personal records). He completes between 5 and 15 repetitions of each exercise with one-minute rest in between sets. He stops a set of exercise earlier if he feels any discomfort in the muscle.\nHe did not report Delayed Onset Muscle Soreness (DOMS) or McArdle symptoms following initial sessions. After three months of resistance training, he found his sleep pattern, overall stamina and McArdle symptoms improved. He is now able to walk two kilometres stopping only once, as opposed to previously when he would have to stop multiple times. He has also found it easier to perform everyday tasks such as changing a car tyre. He has been eating a balanced diet. He autonomously chose not to take any supplements containing glucose pre- or intratraining session. |
A 61-year-old female, known diabetic, hypertensive, asthmatic with past history of resolved stroke and currently on medications for comorbidities and was diagnosed with locally advanced pancreatic cancer (body of pancreas) with metastases to liver and retroperitoneal lymph nodes. She had received palliative chemotherapy as a disease-modifying treatment. She was referred to Palliative Medicine Department for best supportive care in view of her disease progression and declining health.\nShe presented with severe pain in the lower back and left buttock region, radiating to the left lower limb. Pain score was 7–8 out of 10. The pain descriptors suggested mixed nociceptive and neuropathic type pain. It affected her activities of daily living and disturbed her sleep significantly. It resulted in significant distress to both the patient and her caregivers. On examination, she was found to have mass over her left buttock area associated with tenderness. No local changes were found. She was referred to radiotherapy for palliative radiation to left buttock mass region. All her laboratory parameters were within normal range. Analgesia was optimized on a titrated dose of oral morphine 30 mg sustained release tablet twice a day. In addition, adjunct analgesics that included pregabalin and etoricoxib were commenced alongside preemptive side effect-limiting medications. Her pain was well controlled with palliative radiotherapy and opioid analgesics.\nAfter 8 days of regular medications, she presented to outpatient department with a 2-day history of involuntary movements over the right arm with ataxia. On examination, she was conscious and oriented to time, place, and person. Pupils were constricted and sluggishly reactive to light. Right upper arm showed involuntary movements likely to be focal seizures, with decreased palmar grasp of the right hand. All routine laboratory tests were sent and an MRI brain was ordered. She received injection phenytoin sodium as a loading dose for her suspected focal seizures and tablet morphine was stopped immediately. Over a period of 1 h, she became drowsy, minimally responding to painful stimuli. She also developed hypotension with a blood pressure of 94/64 mmHg and hypoventilation with a respiratory rate of 8/min. Pulse rate was 100 beats/min, and saturation on room air was 98%. On examination, her pupils were pinpoint and nonreactive to light. Deep tendon reflexes were also sluggish. She was diagnosed of having opioid-induced neurotoxicity and was immediately given injection naloxone intravenously, starting with bolus of 40 mcg and assessed for response. Bolus was repeated every 2–3 min. The patient became conscious and oriented with five bolus doses (total dose – 200 mcg). Her pupils became normal and were equally reactive to light. Respiratory rate normalized to 18 breaths/min. On chest examination, she was found to have wheeze over the right infrascapular area. She was observed for any signs of recurrence of opioid toxicity for the next 2 h. Meanwhile, she received supportive intravenous fluids for her low blood pressure. Her laboratory investigations revealed acute kidney injury (mainly related to dehydration) with serum creatinine of 2.1 mg/dl. Other parameters were within normal range. Her MRI brain was normal. She was admitted for observation and management of acute kidney injury. After 5 days of treatment, she did not had any signs of recurrence, and her creatinine dropped down to 1.1 mg/dl. She was discharged with good pain control with fentanyl transdermal patch 12.5 mcg/hr, paracetamol, and pregabalin.\nPossible contributors for opioid toxicity in her are acute renal injury, old age, and frail condition. |
A 52-year-old male patient presented with the complaint of sudden vision loss in his left eye 3 days earlier. Past medical history was significant for chronic kidney disease, secondary hypertension, chronic hepatitis C virus infection and arrhythmia. Ophthalmologic examination revealed best corrected visual acuity of 10/10 in the right eye and 4/10 in the left eye from the temporal field. Confrontation test revealed inferonasal visual field loss in the left eye. Direct and indirect light reflexes were normal in both eyes and there was no relative afferent pupillary defect. Anterior segment examination was normal and intraocular pressure was 13 mmHg in both eyes. Dilated fundus exam demonstrated soft exudates consistent with hypertensive retinopathy in the right eye. Fundoscopy of the left eye revealed an area of pallor in the superotemporal quadrant and the macula with macular cherry red spot, which were consistent with occlusion of the superotemporal branch of the left retinal artery (). On OCT, peripapillary retinal nerve fiber layer (RNFL) thickness was within normal limits (). In the patient’s visual field, there was an inferonasal defect in the left eye corresponding to the occluded region (). The patient was treated with a single dose of 500 cc intravenous dextran-40 and 200 mg intravenous pentoxifylline. In etiologic studies, Doppler ultrasonography revealed an atherosclerotic stenosis in the right and left main carotid arteries and a calcified plaque causing luminal narrowing in the left internal carotid artery. Transthoracic echocardiography revealed second- to third-degree aortic valve regurgitation and first-degree tricuspid valve regurgitation. There was no improvement in visual acuity or visual field despite treatment. At follow-up 7 months later, OCT showed thinning of the superior, inferior and temporal peripapillary RNFL (). On the thickness map, ganglion cell layer was thinner in the superior and temporal areas (). Decreased vascular density in the superficial and deep capillary plexus consistent with ischemia in the regions supplied by the superotemporal branch of the retinal artery was observed in a 6x6 mm macular field on OCTA (). The borders of the ischemic area were more clearly seen in en face images (). In optic disc OCTA, capillary density was reduced in the superotemporal region and collateral vessels were present in the area (). When compared to the fellow eye, there was a decrease in the macular deep and superficial capillary density in the superior and temporal quadrants () and a decrease in peripapillary capillary density in the superior quadrant (). Visual field loss persisted in post-treatment threshold perimetry (). |
The patient is an obese, but otherwise healthy, 34-year-old male presenting for a robotic laparoscopic inguinal hernia repair. A preoperative electrocardiogram (EKG) revealed a third-degree atrioventricular (AV) block, which was not recorded in the preceding surgical preoperative note. Our patient knew of his condition but did not disclose this information to anyone until it was discovered via history, physical, and chart review on the day of surgery. He was not aware of the risks associated with the anesthetic procedure and did not have an established cardiologist following his condition. The last echocardiogram was nine years ago in 2011, which revealed no systolic, diastolic, or valvular abnormalities. His last visit to a cardiologist was five years ago in 2015, during which he obtained an EKG unchanged from the normal preoperative EKG he received on the day of his elective surgery. While his EKG on the day of surgery was normal, anesthesiology preoperative history and physical revealed that he has a history of congenital complete AV block. He has no current pacemaker in place and his complete AV block was not followed by a cardiologist when he presented for surgery.\nOur patient's past medical history was significant for bradycardia, chest pain, dyspnea on exertion, hypertension, and complete heart block. However, even though our patient's past medical history was significant for the above, his present medical status and vitals were within normal limits, with the exception of his elevated blood pressure at 169/110. History and physical revealed that he was able to do heavy work around the house, such as lifting furniture and running for exercise, which would classify the patient as having greater than 10 METs. However, even though he was presently asymptomatic with greater than 10 METs, the fact that he had no current establishment of cardiac care warranted further investigation, especially with his concurrent hypertension. A cardiologist was called preoperatively for a consult.\nThe patient was thoroughly counseled on his condition and anesthetic risk. Following a consult with the attending cardiologist, his general surgeon, and the attending anesthesiologist, the decision was affirmed to continue with the elective surgery with the aid of a perioperative temporary venous pacer and central line placement. In most circumstances, cases like this would be deferred, but with the use of a temporary venous pacer, a MET of greater than 10, and a cardiology consult, it was avoided.\nA 6F introducer sheath was inserted into central access via the subclavian vein for transvenous pacing. A 5F balloon-tipped temporary venous pacer catheter (covered in a sterile sleeve) was inserted 20 centimeters into the sheath and then floated under direct fluoroscopy guidance when in the hybrid room. A biopatch was placed at the entry site and an introducer sutured in place. The 5F balloon-tipped pacer catheter with balloon inflated (covered in a sterile sleeve) was advanced using fluoroscopic guidance until it reached the right ventricle apex at approximately 40 centimeters. The balloon was deflated and the capture of pacer function was verified down to 1.5 milliamps as illustrated in Figure [-]. The pacer controller was then placed at 10 milliamps output for intraoperative pacing.\nThe pacer catheter was then secured to the introducer sheath with a suture tie and stabilized to the shoulder. At the end of the procedure, the catheter remained anchored by sutures with a sterile dressing over the entry site. Chest X-ray (CXR) confirmed the placement of the transvenous pacemaker lead in the right ventricle, as demonstrated in Figure [-]. The patient tolerated the procedure well and with no complications. Muscle relaxers used include intravenous (IV) succinylcholine. The anesthetics administered include IV propofol for induction with inhalational sevoflurane to aid in the maintenance of stage III anesthesia. The patient remained intraoperatively stable under general anesthesia, with a pacer wire throughout the surgery without complication. Postoperatively, the anesthesiologist removed the pacemaker and side port. The consulting cardiologist followed up outpatient with 24-hour telemetry monitoring. |
The patient is a 39-year-old African American female who presented with a 3-year history of a tender palpable right lower quadrant abdominal mass. The associated burning pain increased with menses. The patient has a surgical history of an appendectomy and four prior Caesarean sections. Six weeks prior to presentation, she underwent a total laparoscopic hysterectomy with bilateral salpingectomy, as uterine fibroids were suspected to be the potential cause of her pain. Pathology revealed multiple myometrial leiomyomas and benign fallopian tube segments with a 5-mm paratubal cyst. Since the surgery, she reported a sudden increase in size of the lesion and acute worsening of pain. Review of systems revealed only mild constipation. Physical examination demonstrated a 7 cm × 4 cm swelling in the right lower quadrant that was exquisitely tender to palpation and exacerbated by movement. Due to the mass being away from her previous trocar incision and its cyclic nature, an endometrioma was expected.\nComputed tomography (CT) scan of the abdomen and pelvis with contrast revealed a high-density soft tissue opacity measuring 5.8 cm × 4.4 cm in the subcutaneous tissue of the right lower quadrant abutting the anterior aspect of the abdominal wall and associated moderate surrounding subcutaneous soft tissue stranding. These findings were suggestive of a high-density seroma versus post-surgical hematoma. displays the pertinent CT images from admission.\nExcisional biopsy of the anterior abdominal wall mass was performed using a transverse lower abdominal incision over the mass. Dissection was performed around the mass, maintaining a circumferential oncologic margin all the way down to the anterior rectus fascia. The posterior portion of the tumor was found to be invading the anterior rectus fascia, which was excised along with any affected muscle and delivered en bloc to the pathologist. A photograph of the 6.5 × 6.5 × 6 cm mass is shown in . A 15F JP drain was placed in the subcutaneous defect. The patient tolerated the procedure well and was seen in clinic 1 week later with complete resolution of pre-operative pain.\nPermanent pathologic analysis revealed fibroadipose tissue with interspaced endometrial glands and stroma, consistent with endometriosis. No endometriosis was identified at the margins. H&E stain of the mass is shown in . |
A 5-month-old boy was referred to us for further examination of his right upper-eyelid tumor. The tumor was 8 mm in diameter and observed in the upper eyelid (fig. ). According to his parents, the tumor was first noticed at the child's birth. As the patient could open his eye, it was assumed that the tumor most likely would not affect the visual development in his right eye. Therefore, at that time, we decided that a follow-up examination was the best option for the patient. However, due to a gradual enlargement of the tumor, the patient returned 2 years later and underwent further evaluation (fig. ). At the time of the follow-up examination, the tumor exhibited a distinct boundary and was 12 mm in size. Physical examination revealed that there was no pain or any inflammatory reaction in the lid. When observed from the side of the palpebral conjunctiva, the tumor was found to be buried beneath a portion of the tarsal plate and showed a translucent configuration (fig. ). A T2-enhanced magnetic resonance imaging (MRI) examination demonstrated that the tumor was well demarcated from the surrounding tissue. This uniformly high-intensity lesion was 12 mm (height) × 12 mm (width) × 8 mm (depth) in size, with the lesion observed to be in direct contact with the tarsal plate (fig. ). Since the tumor caused blepharoptosis in the patient's right eye, this subsequently led to oblique astigmatism. Therefore, 2 months later, it was decided to surgically remove the mass lesion. The wall of the tumor was easily separated from the surrounding connective tissues, with the exception of the portion where the tarsal plate was attached. As a result, the tumor was completely removed along with a round defect of the tarsal plate (fig. ). Pathological examination showed that the tumor was composed of a thin wall and contained a transparent liquid. The wall consisted of a single cuboidal epithelium that covered the inner luminal surface without decapitation. An area of papillary process proliferation was also observed (fig. ). Based on these findings, the tumor was diagnosed as eccrine hidrocystoma. Although there was obvious improvement in the blepharoptosis after the surgery, his best-corrected visual acuities remained at 0.2 OD and 1.2 OS due to the refractive astigmatic amblyopia that was present at 2 weeks after the procedure. Thus, the patient was provided with a prescription for glasses and, with the help of his parents, began a training program designed for the visual recovery of his right eye. Two years later, his best-corrected visual acuities recovered to 1.0 OD (fig. ). |
A 16-year-old boy with discharge and pain behind the anus was admitted to the hospital. He had sepsis at the same place for approximately 1 year and underwent spontaneously drainaged 3 month ago. Several pilonidal sinus openings were noted in the midline, overlying the coccyx. A lump of tenderness was found left lateral to the openings (). Ultrasound revealed a 3 cm length sinus track beneath the openings which extend to a 2.2 cm × 2.0 cm sepsis left laterally. There was no internal opening to the anal canal.\nThe patient was placed in prone position under spinal anesthesia. One dose of metronidazole was used intravenously during operation. A blunt probe was gently placed through the sinus openings at the midline. The tip of the probe extends to the abscess; tufts of hair were found confirming that this represented pilonidal disease. Anoscopy was then performed and the anal canal was inspected in its entirety. The anal canal was normal; there was no evidence of disease. A 3 cm ellipse incision was made by a blade to remove the skin, subcutaneous tissues, and the pilonidal sinus tract en bloc. A counter incision was made at the extension of sepsis to drain. There was little pus and infected granulation tissue presented in the sinus track, which was submitted to pathological evaluation. The wound was then irrigated with normal saline and hemostasis was obtained. Ten 2-0 silk sutures were put through the 2 incisions and secured loosely to themselves (). A clean, dry, sterile dressing was placed over the top. Patient was given intravenous metronidazole daily for three days postoperatively. Dressings were changed by doctor twice per day at 8 am and 4 pm, respectively. The suture dragging process started from the first day after surgery during dressing change. After irrigation with normal saline, part of the silk sutures which were inside the sinus tract was dragged out and cleaned then turned back into the cavity. All sutures were removed at day 8 postoperatively when the wound became fresh with minimal drainage. Then a 16# straight red rubber catheter with hospital vacuum system was used to assist the wound healing. The top of the catheter was cut into oblique shape and fixed by tape at the edge of the abscess wound. A 5 cm × 5 cm sterile cotton gauze was folded and put at both sides of the wound. Then 3 pieces of sterile gauze were put on the surface of the sinus cavity. A 15 cm × 15 cm antimicrobial incise drape (Ioban 2, 3 M Health Care, MN, USA) covered catheter and gauzes for seal (). The catheter was connected to a hospital vacuum system applied with continuous negative pressure (−30 kPa) to close the wounds. The dressing and catheter were changed and wounds were cleaned every 2 days. The 14# and 12# catheters were used in sequence to correspond to the size of the wound cavity. The vacuum assisted therapy was stopped untill the wound cavity contract noticably 1 week later and patient was discharged. Complete wound reepithelialization was obtained at 22 days postoperatively (). Repeated ultrasound confirmed no track and sepsis at the natal cleft. The patient recovered well and remained asymptomatic for 12 months. |
A 36-year-old male patient with a 6-year history of chronic kidney disease of unknown etiology was brought to the emergency department due to neurological impairment that started during the previous hour characterized by altered mental status while he was walking down the street. On examination he was stuporous, with poor response to external stimuli. The patient was admitted to the hospital and vital signs and capillary glucose were determined. Glucose levels were 20 mg/dl and increased to 42 mg/dl after a 50 ml infusion of 50% dextrose. During his stay his mental status recovered after glucose levels were returned to normal parameters, requiring high doses of intravenous glucose. After his stabilization he was transferred to the internal medicine department. We confirmed that the patient was not taking medications that would cause hypoglycemia. On physical examination he was somnolent and pale, with slight oedema in both legs. A new episode of symptomatic hypoglycemia was observed while he was receiving an infusion of 20% dextrose. The infusion rate at that moment was 10.416 ml/hr. The infusion was not being weaned off or was an acute disruption. It caught our attention that although the patient was on a 20% dextrose infusion, he continued with hypoglycemia. Blood samples were taken and the results were abnormal ().\nAlthough hypoglycemia could be explained by chronic kidney disease, the diagnosis of insulinoma was considered, so a computed axial tomography with double contrast was taken but unfortunately there were no abnormal findings. During the patient's hospital stay he received several treatments that failed to achieve proper glucose control. We used ascending doses of diazoxide up to 600 mg/day with a poor response. In order to reduce episodes of hypoglycemia, we started with low doses of octreotide and found a good response that created tolerance quickly, so we decided to increase the dose by 0.1 mcg/kg/h always observing the same phenomenon. We decided to suspend this treatment when we reached 0.4. mcg/kg/h. Finally, we decided to maintain a continuous infusion of 50% dextrose with which we achieved serum glucose levels between 120 and 160 mg/dl. Magnetic resonance imaging and an endoscopic ultrasonography were performed but no conclusive data on any structural pancreatic disorder were obtained. In order to locate the tumor, we performed selective intra-arterial pancreatic stimulation with hepatic venous sampling at 0, 20, 40, and 60 seconds. High insulin levels were obtained after a selective injection of 0.025mEq/Kg calcium gluconate in the proximal splenic and gastroduodenal arteries () (). With these results, the patient was scheduled for surgery. During the procedure, bimanual palpation of the pancreas was performed, as well as a pancreatic ultrasound in which no tumor could be identified. The body and tail of the pancreas were resected. For two days the patient had an adequate glycemic control but after a couple of days, he presented with hypoglycemia again. The macroscopic pathology report did not show any tumor compatible with insulinoma; however, on microscopic examination pancreatic islets with elongated cells and clear cytoplasm compatible with nesidioblastosis were seen (). Unfortunately, during his stay at the intensive care unit, the patient developed late-onset hospital-acquired pneumonia and, in spite of treatment, he developed sepsis followed by septic shock which ultimately caused his death. |
An 85-year-old man with visual impairment and no psychiatric history presented to the neurological department of the University Hospital Centre of Liège (Belgium). He reported his five-year history of increasing frequency of visual hallucinations and was able to give a detailed and coherent description of his hallucinatory experiences. The patient suffered from retinitis pigmentosa, a degenerative eye disease causing severe vision impairment due to progressive degeneration of the rod photoreceptors in the retina []. At the age of 3 years, he began experiencing progressive and gradual loss of vision. During his adolescence, his peripheral field of vision progressively narrowed (progressive development of "tunnel vision") and he developed hemeralopia, i.e. night vision deterioration by the abolition of rod cells. At the age of 70 he also lost central vision which resulted in complete blindness at the age of 80. The patient reported a positive family history of CBS.\nThis man started experiencing visual hallucinations at the age of 80. Visual hallucinations gradually became more frequent and occurred many times during the day. At the time of the visit, he repeatedly described seeing bilateral visual hallucinations with vivid details. The hallucinations reported by this patient were well formed, ranging from simple flashes or colored background to more complex with the appearance of common faces, objects and bodies of people, or landscapes. The hallucinations varied in size and color, and were binocular, covering the entire visual field. However, animations (i.e. scenes in motion) were only present in the right visual field. The visual hallucinations were always perceived as pleasant; they generally occurred with the eyes open and did not disappear when closing the eyes and were never accompanied by abnormal perception in any other sensory modality. The patient was fully aware of their unreal nature but he was not able to consciously control their occurrence or content. Based on his clinical history and the diagnostic exams he underwent, a diagnosis of CBS was made by the neurologist. Indeed, the patient fulfilled the four diagnostic criteria for CBS: (1) hallucinations must be complex, repetitive, and persistent; (2) awareness that the hallucinations are not real; (3) no additional delusions; and (4) absence of additional hallucinations in the other senses [].\nThe patient underwent a neuropsychological examination, including the Mattis Dementia Rating Scale [] and the version for the blind (MoCA-BLIND) [] of the Montreal Cognitive Assessment (MoCA) []. Considering his visual impairments, all these cognitive test materials were administered verbally, thereby omitting all vision-specific items.\nThe patient underwent an ophthalmologic examination with, notably, measurements of visual acuity and visual evoked potentials. |
A 17-year-old girl reported to the Department of Periodontics with the chief complaint of unesthetic appearance of her front lower teeth []. On examination, it was found that 6 mm deep and 4 mm wide class II gingival recession (Miller, 1985) was there on the lower left central incisor [Figures and ]. The tooth was slightly labially placed and patient also gave the history of tooth brush trauma. The vestibular depth and the width of attached gingival were also inadequate in the region. There was no mobility associated with the tooth. For the root coverage, increase in width of attached gingiva and vestibular deepening the periodontal plastic surgery was planned with a single stage fenestration technique and root coverage using the periosteal pedicle graft. The patient was advised for the treatment of the isolated gingival recession defect. The patient was in good systemic health with no contraindications for periodontal surgery. She was explained about the surgery and signed informed consent was taken by the patient. A general assessment of the patient was made through her history, clinical examination and routine laboratory investigations. Before surgery, the patient received phase-I therapy, which included oral hygiene instructions and scaling and root planning with ultrasonic and hand instruments. Two weeks after phase I therapy, the patient was planned for surgical procedures. On the day of surgery, local anesthesia was first administered bilaterally by using a mental nerve block. A horizontal incision was made using a no. 15 surgical blade at the mucogingival junction retaining all of the attached gingiva [Figures and ]. A split thickness flap was reflected sharply, dissecting muscle fibers and tissue from the periosteum. This was then sutured in the depth of the vestibule using resorbable 5-0 sutures []. A strip of periosteum was then removed at the level of the mucogingival junction, causing a periosteal fenestration exposing the bone. The care was taken not to remove the periosteal strip completely and to leave it pedicled to the bone and the rest of the surrounding periosteum at the lateral end [Figures and ]. The recipient site preparation included two horizontal incisions. First, intracrevicular incision and a second incision made parallel and apical to the first incision []. The incisions were followed by split-thickness dissection of the facially located tissue up to the level of the vestibular incision so as to create a tunnel [Figures and ]. The exposed root surface was root planed with curettes to remove bacterial contamination and was biomodified using the tetracycline powder mixed with saline. The pedicled periosteal donor tissue was then moved vertically towards the recession area, passing through the tunnel [Figures -]. At repositioning, the osteoperiosteal portion was closely adapted to the recipient site by pressing for 3 min and then sutured along with the overlying gingival tissue, to the recipient bed, using 5-0 resorbable sutures [Figures and ].\nPeriodontal dressing (Coe-Pak; GC America Inc.) was applied over the operated area covering the exposed bone []. Antibiotic therapy (amoxicillin 500 mg, Thrice daily and analgesic (ibuprofen 400 mg twice daily) was prescribed for 5 days. Tooth-brushing was discontinued for the first 2 weeks at the surgical site and 0.2% chlorhexidine mouth rinse was instructed till 4 weeks after surgery. Coepak was removed 10 days after the surgical procedure and the patient was asked to maintain meticulous oral hygiene. Healing had proceeded uneventfully, with secondary wound closure []. In 3 weeks, healing was nearly complete, with minimal post-operative discomfort to the patient []. At 6 months post-operative, root coverage was nearly 100% of the recipient site, with minimal probing depths, no inflammation, and a favorable esthetic result []. |
A 73-year-old male was referred to our hospital presenting with bilateral vocal fold palsy and a rapidly growing anterior neck mass. We performed ultrasound sonography, revealing a bulky tumor in the thyroid gland and multiple bilateral cervical lymphadenopathies. We performed fine-needle aspiration cytology from both the thyroid tumor and the cervical lymph node, both of which revealed SCC. We found no primary tumor in the pharynx, larynx, or esophagus with laryngoscopy and upper gastrointestinal endoscopy, nor could we discover any primary tumor in the trachea using bronchoscopy. We discovered a bulky thyroid tumor and bilateral cervical lymph nodes involved in the common carotid artery and brachiocephalic artery using computed tomography (CT) with contrast effect (Fig. ). Using FDG-positron emission tomography/CT, we found FDG uptake in the thyroid, the bilateral cervical lymph nodes, the lungs, the mediastinal lymph nodes, and the vertebra (Fig. ). We diagnosed the patient with stage IVC primary SCC in the thyroid and initiated treatment with weekly paclitaxel. He received chemotherapy weekly at 90 mg/m2 according to a 6 weeks on/2 weeks off schedule.\nFour months after the initiation of treatment, the objective response obtained from CT presented a stable disease. However, a slightly enlarged tumor resulted in dysphagia and aspiration pneumonia, which led to the patient's death. We performed an autopsy according to the patient's wishes. On a macroscopic level, we found that the greater part of the thyroid was replaced by carcinoma. The tumor and the cervical lymph node metastases involved the common carotid artery and the brachiocephalic artery, but not the trachea or larynx (Fig. ). The tumor had slightly invaded the external muscle layer of the esophagus; the mucosa of the esophagus, however, was intact. We observed multiple tumor nodules in the bilateral lungs and mediastinal lymphadenopathies. These macroscopic findings indicated primary thyroid cancer with distant metastases.\nOn examining the histopathology of all of the carcinomas in the thyroid, cervical lymph nodes, and lungs, we observed a palisade arrangement, intercellular bridges, and keratinization with a cancer pearl (Fig. ). We interpreted these histopathological features as pure highly differentiated SCC. Consequently, only in the thyroid was primary SCC proven and never in any other organ. We performed immunohistochemical examinations of the primary thyroid cancer, the cervical lymph node metastases, and the lung metastases; all of which were negative for TTF-1, Tg, and PAX8 (Fig. ). |
A medically free 14-year-old male who was obese sustained an injury to his left knee after jumping from 3 stairs. The patient mentioned that he predominantly landed on his left lower limb with his left knee in full extension and in external rotation. The patient started complaining of left knee pain limiting his range of motion and ability to bear weight immediately after the fall. He was brought to the Emergency Department (ER) of King Saud Medical City (KSMC) by his parents immediately after the injury. On physical examination, the left knee was profoundly swollen and bruised. There was tenderness over the tibial tuberosity and lateral joint line. He was unable to actively move the knee joint. The passive range of motion was painful. There were no signs indicating compartment syndrome or neurological or vascular injury. X-ray radiographs revealed a Watson-Jones type IIIB avulsion fracture of the tibial tuberosity apophysis (). A CT scan showed a step of the articular surface more than 2 mm extending to the posterior-lateral epiphyseal part of the proximal tibia (lateral tibial plateau) ().\nThe patient was admitted and was prepared for operative management. A procedure was planned and done on a radiolucent table under general anesthesia. A tourniquet was used to avoid excessive bleeding during the procedure. The tourniquet was inflated after pulling down the quadriceps to avoid blocking the reduction due to the extensor mechanism. The leg is prepped and draped according to the standard orthopedic protocol. Anterolateral approach of the knee was used with an incision starting from the lateral upper border of the patella to 10 cm down. Deep fascia was opened anterior to the iliotibial tract. The fracture line was identified; the reduction of the articular step was done using a reduction clamp and assured using a portable image intensifier on flexion and extension of the knee without arthrotomy. We avoided arthrotomy of the joint to not make it vulnerable to infection and possible scarring. Stabilization of the reduction was maintained using a k-wire. Definitive fixation was achieved with three 3.5 mm partially threaded cancellous screws placed under fluoroscopic guidance for the tibial tuberosity fracture. A proximal tibial plate was slid laterally and was used to buttress the lateral tibial column. Careful placement of the screws was done to not cross the physis with the help of a C-arm (). After fixation, good hemostasis was achieved, drain was placed, and the range of motion was assessed which was full. Closure was done layer by layer, then dressing after. The postoperative plan was to immobilize the knee in a cylindrical cast for 3 weeks with no weight bearing on the left lower limb with the use of crutches for ambulation.\nPostoperative knee CT scan is requested to ensure that the fracture is anatomically reduced. The patient received analgesia and antibiotics, and drain was removed 24 hours post-op. The patient was seen in an orthopedic clinic after 3 weeks, there were no signs of surgical site infection, and the clips were removed. The controlled range of motion was advised using a hinged knee brace throughout the day for 4 weeks. A follow-up X-ray () shows that the fracture is aligned with no loss of reduction or displacement. Physiotherapy is advised 7 weeks postoperative management focusing on the range of motion and strengthening. The patient was seen 6 weeks later, he had full range of motion with no deformity, and there were no complaints reported by the patient like locking or pain. |
A 15-year-old male patient was admitted to pediatric neurology outpatient clinic for the complaint of abnormal posturing. Developmental history revealed that he had been diagnosed with severe mental retardation in early childhood. He had marked delays in basic motor skills, never begun speaking and has been receiving special education for six years. He attended a child and adolescent psychiatry outpatient clinic four years ago for hyper-activity, irritability, aggressiveness, and self-injurious behavior. With the diagnoses of attention deficit hyper-activity disorder, conduct disorder and mental retardation, he has been on risperidone 2 mg/day for the last four years. Approximately one month ago, the patient gradually developed tonic flexion of trunk and head toward left. With the suspect of an adverse effect, medical records were carefully reviewed for a detailed medication history. The patient had used no psychotropic medication other than risperidone through these four years. No antiemetic use was also found. Medical records revealed the use of antibiotics and antipyretics 3–4 times a year for colds, upper respiratory tract infections, and bronchitis. No movement and neurodegenerative disorder were reported in family history.\nNeurologic examination showed that the patient had clear consciousness, with no abnormalities in sensation of the extremities, cranial nerve functions, muscular strength and deep tendon reflexes. He could not speak, but he could follow the orders. Tonic flexion of trunk and head to the left and shift of the center of gravity toward the left were observed (). He did not have any history of dystonic movements and no evidence of other extrapyramidal symptoms was found. Common blood and biochemical tests were in normal range. Magnetic resonance imaging (MRI) of the brain detected global cortical atrophy, agenesis of the corpus callosum and no abnormality in basal ganglia.\nDue to the emergence of abnormal posture after long-term risperidone use, the diagnosis of PS was suspected. Possible neurological diseases were excluded by family history, neurological examination, laboratory tests and neuroimaging. Risperidone dose was decreased to 1 mg/day based on the recommendation of child and adolescent psychiatry department. Two weeks following the dose reduction, abnormal posture and flexion of the trunk and head disappeared. Hence, the diagnosis of risperidone induced PS has been verified (). |
A 24-year-old South Asian woman of Punjabi ethnicity presented with fleshy mass protruding through midline vertical abdominal scar and bleeding from the mass during menstruation for the past 5 months. She was primigravida; she underwent a caesarean section 6 months earlier at term gestation for breech presentation in a local hospital. She delivered a normal healthy baby boy and the immediate postpartum period was uneventful. On the sixth postoperative day she noticed serosanguinous discharge from her abdominal wound and wound dehiscence was diagnosed. She was managed conservatively and the wound was left for secondary healing by the attending physician. Her history revealed that she was given antibiotic coverage during this time. The major portion of her wound healed in 1 month leaving a 2 cm area in the middle of her vertical scar. An investigation at the time of her caesarean section revealed that she was anemic (hemoglobin 8 gm %). Peripheral blood film revealed microcytic hypochromic anemia. On admission to our institute she was emaciated, thin built, anemic and her vital signs were normal. An abdominal examination revealed a 2×2 cm fleshy mass protruding through the middle part of her infraumbilical abdominal scar. On per vaginal examination her uterus seemed attached to the anterior abdominal wall at the scar site and with cervical movement the mass was getting retracted into her abdomen. With her history of menstruation through the abdominal wound a provisional diagnosis of scar endometriosis/uterocutaneous fistula was made and ultrasonography (USG) and fistulogram were suggested. USG showed normal uterus and adnexa and fistulogram showed communication with intraperitoneal cavity. A tissue biopsy revealed granuloma. She was planned for excision of the fistula tract and repair. On an operating table methylene blue dye was injected through the wound to mark the fistulous tract and dye was found to be escaping through her vagina confirming communication with uterine cavity. An elliptical incision was made around the fleshy mass and on entering the abdominal cavity, the left side of her uterus was adherent with her anterior abdominal wall at the scar site and the fimbrial end of her left fallopian tube was found to be protruding through the abdominal scar. A probe (dilator) was passed through the tube and diagnosis was confirmed (Fig. ). The tube was pulled inside and adhesiolysis of uterine adhesions from anterior abdominal wall was done. The scar edges were freshened and the incision was closed in layers. She had an uneventful recovery. She was followed up monthly for 3 months and had no complaints. |
A female patient aged 32 years reported to the Department of Conservative Dentistry and Endodontics, with the chief complaint of pain in the upper anterior tooth. Examination revealed a palatally placed upper permanent right lateral incisor, buccally positioned relative to an upper retained deciduous canine tooth. The upper right central incisor was tender on percussion and presented with a facial groove with a probing depth of 1.0 mm (). Vitality test was negative. The patient was advised a radiograph of the area in question, which showed an impacted permanent canine, and loss of lamina dura and slight amount of radiolucency in relation to the periapical area of the upper right central incisor (). There was an additional root present with the central incisor. Since the deciduous tooth was firm, root canal treatment of the central incisor, extraction of impacted canine and orthodontic correction of palatally placed lateral incisor was advised but the patient refused to undergo any surgical treatment. A CBCT was advised to rule out the association of the impacted canine with the central incisor and to identify the exact position of the additional root of central incisor.\nThe CBCT ruled out the association of impacted canine with the central incisor () and showed a periapical lesion related to both the roots of the central incisor (). Contrary to our expectation in relation to the additional root being positioned in the middle of the labiolingual length of the tooth, we found it to be positioned more facially (). A three dimensional image of the tooth is presented in Figure ( and . A nonsurgical endodontic therapy was decided after discussion with the patient.\nAfter rubber dam application, endodontic access was gained. Two canal orifices (mesial and distal) were located with an endodontic explorer (DG-16, Dentsply Instruments, Surrey, UK). The working length of both canals was determined radiographically () and confirmed with an apex locator (Propex II, Dentsply international, Surrey, UK). The root canals were cleaned and shaped using rotary Protaper files (Dentsply International, Surrey, UK). The mesial canal was instrumented to #F4 and distal canal to #F3. The root canals were copiously irrigated with 2.5% sodium hypochlorite solution. Calcium hydroxide/distilled water paste was then placed as an intracanal medicament. After 2 weeks, the tooth was asymptomatic and the root canals were obturated with corresponding Protaper gutta-percha and AH Plus sealer (Dentsply De Trey, Germany) (). During sealing of the endodontic access cavity, the patient was given the option of restoring the facial groove with composite resin, which she refused. One-year follow-up radiograph presented satisfactory healing (). The patient was absolutely asymptomatic and satisfied with the treatment outcome. |
A 42-year-old woman with a severe lower abdominal and pelvic pain due to advanced cervical uterine cancer was referred to our pain clinic. Cancer had been diagnosed five months ago. The patient had used many pain killers and the pain was intractable to many analgesic modalities. She was a candidate for a superior hypogastric plexus block. In a uterine cervix biopsy, moderately differentiated non-keratinizing squamous cell carcinoma was reported. She had five sessions of chemotherapy and 25 sessions of radiotherapy in the past three months. She was receiving methadone 5 - 7.5 mg every eight hours, gabapentin, and acetaminophen for pain management during these three months. This medical treatment was administered by the palliative care service but in spite of this medical management, she was suffering from severe pain in lower abdominal and pelvic areas. Then, she was referred to our pain clinic. The pain was evaluated by the Brief Pain Inventory and the overall pain intensity was 8 out of 10 on the 11-point NRS scale ().\nIn an MRI study, a heterogenous enhancing lobulated mass (9 × 8.5 cm) was reported involving lower uterine cervix and upper vagina suggestive for a cervical tumoral lesion. There were numerous enhancing masses (adenopathies) in both iliac and para-aortic regions, as well. Moreover, enhancing adnexal masses (4 - 5 cm) were observed in the right and left sides. The liver, spleen, pancreas, and both kidneys did not show any mass or abnormality.\nOn the physical exam, she had severe deep tenderness and pain in the lower abdominal and pelvic areas. Organomegaly was felt in the lower abdomen. She did not have any abdominal rebound tenderness; therefore, peritonitis was ruled out. There were radiotherapy-induced blisters on the lumbar skin. A dermatological consultation was requested. No infection or important lesion was reported over the skin of the lumbar area.\nThe origin of the pain was a pelvic malignancy. Consequently, we decided to perform the superior hypogastric plexus block to relieve the pain of the patient. The most disturbing position for this patient was the prone position due to severe abdominal and pelvic pain. The patient could not tolerate this position even for a few moments.\nThe patient was unable to tolerate the prone position. Therefore, we decided to perform the block in the left lateral position (). After positioning the patient and applying vital sign monitoring (SPO2, NIBP, ECG, RR, and HR), midazolam 1 mg and fentanyl 50 µg were administered for sedation. The skin was prepped with povidone-iodine and then draped. Then, 10 mL of lidocaine 0.5% was infiltrated for skin local anesthesia. Two Chiba needles (15 cm, 20 G) were bilaterally placed 6 cm lateral to the midline at the level of the L5 vertebra. Then, the needles were advanced from the lateral to midline direction based on the tunneling technique and oblique views ( and ) obtained by the guide of the C-arm fluoroscopy. The target point was the anterolateral surface of the L5-S1 junction. We placed the right needle first and subsequently put the left needle (). All steps of the procedure were done under the guidance of the multiple fluoroscopic images (oblique, lateral and anteroposterior views).\nAfter the injection of the contrast, the right position of the tip of the needles was confirmed. The contrast was not adequately distributed toward the anterior surface of the S1 and did not perfectly cover the L5 - S1 junction. Bilateral superior hypogastric plexus was blocked in the lateral position with 10 ml of bupivacaine 0.25% and 40 milligrams methylprednisolone on each side ( - ).\nThe patient was comfortable in the lateral position during the procedure. The images were accurate and the block was performed straight forward without any technical problem. The pain score reduced to 2 out of 10 on the 11-point NRS scale after the end of the procedure. |
A 42-year-old Caucasian woman was referred for the evaluation of two bilateral asymptomatic, cystic lesions in the mandible. It was an incidental radiograph finding of unknown first appearance and evolution. Intraoral examination revealed clinically absent third molar teeth with no swelling or tenderness []. The medical history was nonsignificant and there were no association with syndromes.\nIn cone beam computed tomography (CBCT), it was shown two well-defined unilocular radiolucent areas surrounded by thin sclerotic border related to unerupted mandibular molars. The one on the mandible right side showed diameter of approximately 23.64 mm and almost reached the inferior border and the left one 16.57 mm. Both third molars were displaced, the right one more grossly and apparently involving the distal root of the second molar and were located intimately with the mandibular canal [].\nSurgical marsupialization and aspirative punctuation of the bigger lesion on the mandible right side was performed to attempt a bone formation. Three months later, no relevant new bone formation was observed. Therefore, surgical enucleation of both cysts was selected as treatment. Routine blood and urine tests were advised before the surgery and the results were within the normal limits. Under general anesthesia, the two cysts were enucleated together with the excision of the associated third molars and due to the lesion size and localization it was placed a plate for fixation to avoid postoperative fracture [].\nThe histopathological examination of the left lesion revealed a cyst cavity lined by nonkeratinized, stratified, squamous epithelium of varying thickness []. Underlying, in the cyst wall, sparse mononuclear inflammatory cells were observed. In addition, the right lesion showed epithelial hyperplasia of the cyst lining and capsular connective tissue with areas of intense chronic inflammation []. Based on clinical, radiographic, and histopathological features the final diagnosis of dentigerous cyst was confirmed in both lesions. Now-a-days, the patient is on 18 months radiograph follow-up with favorable osseous formation with no evidence of recurrence of the cysts []. |
The patient was a 65-year-old woman without notable antecedents presented to our institution for progressive left hip pain for approximately 8 months. It was a mechanical pain of the hip well relieved by the usual analgesics. The appearance of walking distance and the poor response to analgesics forced her to consult in our center.\nThe BMI was 35,5. The walk was almost normal. There was no cutaneous scar on the lateral side of the left hip or on the ipsilateral buttock. There was a good trophicity of the abductors. Lateral rotation and abduction were markedly diminished. The rest of the exam was strictly normal. The pelvis AP () and lateral () left hip radiographs revealed signs of hip osteoarthritis. We concluded that it was a symptomatic left hip osteoarthritis that was more and more disabling in an obese woman of 71 years with no particular history. We indicated THA by posterolateral approach.\nIn the operating room, after the skin incision and subcutaneous haemostasis, we discovered in the adipose tissue about 5 cm thick a kind of well-circumscribed shell of about 2.5 cm of axis. Her incision gave rise to a whitish, thick color, looks a little oily collection (), resembling a purulent collection (). A sample for bacteriological investigation in a lab was carried out. The hull with its clear boundaries within the gluteal fat was resected and entrusted to the pathologist. All the neighborhood tissues were healthy (very localized lesion).\nIn front of this collection which appeared to be purulent, we limited ourselves to the resection of this hull, the cleaning of the wound, and the deferred implantation of the prosthesis.\nCytobacteriological examination of the specimen revealed its greasy appearance, epithelial and lymphocytic cells; there were no visible germs. Histological examination of the resected shell revealed a fibrous wall with chronic inflammatory remodeling made of lymphocytes and plasma cells with no necrosis centers.\nIn the light of these laboratory results, we conducted the interview of the patient, who reported a notion of malaria for about two months to the screen treated with an intramuscular injection on the right buttock of the compounds derived from artemisinin. We found the result of the thick drop before the injection which was positive and that of the injection which had not been negated; the patient was then successfully treated orally. The sample was sent to a lab for confirmation by artemether identification by thin layer chromatography (TLC).\nA sample of 40 g of human fat was treated with ethyl acetate (50 ml × 3) after filtration on Whatman paper, the solvent was evaporated, and the residue was taken up with acetone (40 ml) constituting the sample to be analyzed. Artemether was purchased from a local pharmaceutical company.\nImplementation of the TLC: solution to be analyzed: 20 μl of sample; control: artemether (80 mg/mL), 10 μl deposit; support: silica gel GF254; mobile phase (10 ml): dichloromethane, ethyl acetate (7/3); and developer: 25 ml anisaldehyde reagent, 5 ml concentrated acetic acid, 450 ml ethanol, and 25 ml concentrated sulfuric acid. Using a capillary tube, 20 μl of the sample was deposited on the plate (silica gel GF254), the control 10 μl. The plate is placed in a tank previously saturated with the migration or elution solvent (mobile phase) which covers the bottom of the tank at 5 mm height. The migration of eluting solvent causes the substances contained in the samples at various speeds; spots are formed characterizing the substances present in the sample.\nThe plate was removed from the tank as soon as the solvent front reached about 9 cm. The plate was dried and observed under a UV lamp at 254 nm and then revealed with the developer which will characterize the artemether in human fat.\nThe plate then shows an orange spot on the left side of the sample and a spot with the same color on the right side of the control; the two spots have the same front report as shown in . This indicates that there was artemether in this human fat sample. |
A 12-year-old boy was brought to our policlinic by his family with a complaint about weakness in his right leg and inability to walk. His family told us that it was caused by the firearm injury which had occurred two months before.\nOn physical examination, it was found that the patient had incision scars left as a result of surgical intervention made two months before to take out the buck shots in medial and posterior regions of the thigh after the firearm injury. It was written in his surgical report that he had sciatic nerve injury and the buck shots in the body had been taken out. Sense and reflex examination could not be made because of strong pain. The intensity of pain was measured by visual analogue scale (VAS) ranging from 0 to 10 and values were 10 and on a scale for assessment of neuropathic symptoms and signs (LANSS) were 22 before treatment. Muscle-power examination could not be made, either, because of the strong pain. However, as far as it was evaluated there was no active movement in right foot: dorsiflexors and plantar-flexors. In evaluating the range of motion, a goniometric measurement was taken with minimal contact to the patient due to severe pain, and the right knee was measured in 105-degree limited flexion (75-degree extension was needed for complete extension) and the right foot ankle in 20-degree limited plantar flexion (20 degrees was needed for neuter position of foot ankle). The patient was accepted to our clinic for rehabilitation. As it is planned to give medicine to the patient, it is aimed to determine whether the values in liver and kidney were normal and there was infection and diabetes or not, so the blood analyses of the patient were asked. AST, ALT, fast blood, glucose, creatine, hemogram sedimentation, and C-reactive protein (CRP) tests were found to be normal.\nThere was no pathologic substrate in X-rays of right knee and ankle image explaining joint movement limitation and severe pain. Electroneuromyography examination on the patient could not be evaluated definitely because of the pain; however, it was determined that there was nearly a total partial axonal degeneration which was heavier in the right sciatic nerve peroneal division and lighter in tibial division. Soft tissue ultrasound of the gluteal region; venous and arterial doppler ultrasound of right lower extremity, were normal.\nAs the patient had to keep his knee in flexion position due to severe pain and movement limitation, and his X-ray and ultrasound images were normal, CT and MRI were not asked. This pain in patient was evaluated as allodynia. A rehabilitation programme (stretching, increasing the range of motion, proprioception, strength exercises, TENS, and surface heaters) was planned for the patient but could not be applied because of severe allodynia. As the rehabilitation programme could not be implemented, we consulted the paediatric neurologists and acted on their suggestions. Thus, topical and oral nonsteroidal anti-inflammatory medication according to the patient's age was given to the patient for ten days. As there was no decline in complaints and the rehabilitation programme could not be implemented, daily 10 mg/kg dose gabapentin was given to the patient by consulting paediatric neurology. Regarding patient complaints, the dose of medicine was increased up to 16 mg/kg a day. Beginning from the first week, the patient's neuropathic pain complaints declined; thus, a rehabilitation programme could be implemented. The patient's value of VAS and LANSS was 3 and 0, respectively, in the second week. Apart from light dizziness for the first 1-2 days, there were no side effects during gabapentin treatment. In the twentieth séance after the start of gabapentin treatment; flexion and extension of right knee, right foot ankle plantar flexion was unrestricted, and right ankle dorsiflexion was measured as 100 degrees. Foot plantar flexion and dorsiflexion were 3/5, and right knee flexion, extension, right hip flexion, extension, abduction, and abduction of muscle powers were 5/5 level. The patient was discharged and given home exercises programme. The medicine was lessened gradually to the end of the second month. The patient came for monthly control each month, and he did not have any additional complaints. |
52-year-old man, plumber by profession, presented at the emergency department with one day history of having sudden onset spontaneous pain and functional impairment of left knee while he was walking. The patient had history of hypertension. He had no history of taking medications such as corticosteroids or fluoroquinolones and did not remember having suffered any knee pain previously. On clinical examination effusion was present in knee. It was possible to palpate a gap between the distal patellar tendon and the tibial tuberosity and patient was not able to extend the leg. X-ray of the left knee showed a high patella and presence of calcification in the distal part of the patellar tendon (). Ultrasound later confirmed the diagnosis of distal avulsion of the patellar tendon. Although CT scan and MRI are superior modalities for confirmation of the diagnosis but based on our clinical examination, typical radiographic finding and ultrasound imaging, we considered these expensive investigations unnecessary in this case. On the second day, patient was taken to the operating room. Under tourniquet control, patellar tendon was exposed through anterior approach to the knee. Distal insertion of the tendon was found to be completely avulsed from the tibial tuberosity with some flakes of sclerotic bone (). The torn tendon was found to have degenerative changes at the injury site. The tendon stump was debrided and refashioned. Locking double Krackow stitch was taken with FiberWire number 5, and then it was anchored to tibial tuberosity after drilling four 2.0 mm transverse holes in tuberosity. Full range of motion of the knee was performed to evaluate the repair and we found it satisfactory. Repair of paratenon was performed and wound was closed. Postoperatively, knee was locked in a brace at 0 degrees of extension and the patient was encouraged to do isometric quadriceps strengthening exercises. Patient was allowed weight bearing as tolerated. A range of motion exercises were progressively started after 5-week follow-up. The patient achieved full active range of motion by third month of follow-up, complaining only of a slight tenderness on pressure at the level of the tibial tuberosity. X-rays were taken in the last follow-up at 5 months showing normal position of the patella (), while clinical examination revealed full recovery. |
A 66-year-old retired married, male patient was seen in the pain clinic in mid-2014 complaining of classic episodic cluster headaches for the previous 10 years. During this time, he had pain-free periods lasting for about 6 months. Headaches occurred up to 8 times a day, starting as a short stabbing pain in the medial aspect of his right eye and gradually spreading to the entire right side of his face, vertex and upper back of the head. The pain attacks would last between 45 and 75 min; he was completely pain-free between attacks. The pain felt ‘like his head was going to explode’ and was unbearable. The pain was associated with rhinorrhea in the right nostril and obvious conjunctival injection in the right eye without oedema. The patient was extremely restless and absolutely nothing he could do would relieve the pain. He was treated with a greater occipital nerve block comprising 40 mg methylprednisolone and 20 mg bupivacaine. The nerve block afforded him complete pain relief within minutes and lasted for 12 months. The block was repeated on two occasions, each time providing relief for a further 6 months. Subsequently, the frequency and intensity of the headaches subsided and by mid-2016 he was free of any headache for the next 2.5 years. Apart from a past history of an abdominal aortic aneurysm which was operated on, he was otherwise healthy. His family history was unremarkable.\nThe patient presented to the pain clinic again in July 2019 after suffering for 6 months from a new constant stabbing pain radiating from the back of the right side of his head to the right side of both the vertex and forehead, right ear and towards the jaw. Numbness and paresthesias were also present in this distribution. He had no neck pain. In addition, the intermittent tearing and conjunctival injection of the right eye that he previously had with his cluster headaches had become continuous and the eyelid was also edematous. He had excessive rhinorrhea. He often felt restless, particularly at night. The basal Numerical Rating Scale Pain Score (NRS) was 6 out of 10. Pain exacerbations that reached an NRS score of 9 out of 10, occurred 2–3 times a day and lasted 20–30 min. They were occasionally exacerbated by neck movements.\nOn physical examination, his right eye was mildly teary, with conjunctival injection and slight oedema of the eyelid. He had hyperesthesia over the right vertex and right side of his forehead. The neurological examination was otherwise normal. Passive extension of the neck was extremely limited and reproduced the severe exacerbations of his pain. Palpation of the right SCM muscle not only elicited tenderness but exacerbated the referred pain. This raised the suspicion that the SCM muscle was a source of the headache. Therefore, 30 mg of 1% lidocaine was injected into the right SCM muscle. The intention was to perform a diagnostic injection with a short-acting local anaesthetic to determine if the pain arose from the SCM. No other muscles were injected. Within 4 min of the injection, the severity of his headache decreased from a pain score of 7 out of 10 to 0. The numbness across his forehead and vertex disappeared. Neck range of motion became painless and much improved. The right-sided tearing and conjunctival injection began to subside, followed later by the resolving eyelid oedema. Relief of symptoms was complete for 4 weeks and then partially relapsed.\nHe returned to the pain clinic in September 2019 and, as he requested, received another injection. The right SCM muscle was just as tender as it was in July and palpation of it exacerbated the pain. This time the muscle was injected with 20 mg lidocaine and 10 mg bupivacaine. His pain completely resolved for a further 2 months.\nIn mid-December, when the headache relapsed, a second specialist in both neurology and pain relief medicine recommended that the patient start on a low dose of indomethacin, 25 mg three times a day, and then increase the dose slowly. Co-interventions, including further injections, were withheld in order to determine the efficacy of the indomethacin and so as not to undermine the diagnosis of hemicrania continua. The pain was relieved within 24 h of the indomethacin trial, confirming the diagnosis of hemicrania continua. There was no need to increase the dose, as his headache was eliminated. It was decided to discontinue the indomethacin due to the onset of severe gastric side effects. Six days later, by which time the headaches had completely relapsed, the patient was then given another injection of 20 mg lidocaine and 10 mg bupivacaine into the right SCM muscle. Once again, his pain was eliminated. During visits to the pain clinic for unrelated low back pain in June and October 2020, and February 2021, he reported that both his continuous headache as well as his cluster headaches were still in complete remission.\nOur patient much preferred to have periodic injections than suffer from indomethacin-related side effects. The injections he received had no side effects and afforded him lasting relief. |
A 36-year-old woman with bilateral anophthalmia and hyperparathyroidism in the course of a parathyroid adenoma and nephrolithiasis complicated by renal failure (G-3) with GFR 53 ml/min/1.73 m2, was admitted to an Orthopaedic Department on account of fractures to the shaft of the right femur and the left patella being the result of a fall. Surgical parathyroidectomy had been performed 2 weeks prior to trauma.\nThe patient was in a good state overall, though reporting severe pain in the right hip that prevented active movement, mild swelling of the thigh, and swelling of the left knee with pain in the left patella on palpation. There was no compromising of blood-flow, sensation or motor-nerve function.\nUnder X-ray a pathological fracture of the proximal third of the femoral shaft was revealed, in the place of an osteolytic lesion of diameter 60 mm, with thinning of the cortical layer (). In turn, the second pathological fracture involved the left patella visibly changed by multiple osteolytic lesions (). Further multiple osteolytic lesions were in fact found on the left tibia and the left pubic and sciatic bone.\nGiven the history of parathyroid adenoma, disseminated brown tumours were suspected. The patient was scheduled for surgical treatment of the femoral shaft fracture, and, given the lack of displacement and the extensive bone osteolysis, also for conservative treatment of the patella fracture. An inferior vena cava filter was implanted prior to surgery, given a history of inferior vena cava and bilateral common iliac vein thrombosis.\nThe procedure performed was closed reduction and internal fixation (CRIF) with a proximal femoral nail. A tissue sample was obtained for histopathology, the results sustaining the initial diagnosis of pathological fracture reflecting the presence of a brown tumour.\nAs the surgery was not followed by further complications, the patient was discharged home in a good overall state. And, as she did not attend for initial after-surgery checks, the first such check of an orthopaedic nature was performed one year on from the operation. A CT-scan and X-Rays then showed full bone union at both fracture sites, with no secondary displacement, and with brown-tumour regression seen to be taking place in the lower limbs and pelvis (, ). The patient reported no pain and was able to walk with the assistance of another person. She is now checked regularly by an endocrinologist, and, 2 years after parathyroidectomy, there is no sign of a relapse into hyperparathyroidism. |
A full-term girl, the first child of a 24-year-old mother was born with large skin, muscle, bone and dural defect in the lumbo-sacral area. The patient weighed 2800 g. The patient was evaluated by the pediatric clinic and no other congenital malformations were found. There was no congenital malformation in the family history. Physical examination revealed no neurological deficits and the head circumference was 34 cm. The lesion showed 8 × 5 cm skin defect, covered with transparent arachnoid membrane. Underneath nerve tissue of the spinal cord was split by a perpendicular bony spur and connected from its tip to the upper lamina [Figure and ]. Dura matter was seen on both sides of the lesion extending laterally over the paraspinal muscles. Common vascularization between the skin and the central neural tissue was seen. Brain computed tomography (CT) scan showed mild ventriculomegaly without signs of increased intracranial pressure. Spinal X-rays showed a bony spur on the L2 vertebral column and laminar defect in the lumbo-sacral area []. Under general anesthesia and prone position, the arachnoid layer was removed and the split spinal cord was dissected from the bony spur. There was no neural placode and the spinal cord was found with full neurulation. The spur was seen extending from the posterior border of the vertebral body and penetrating the anterior dura []. However, dural folds around the base of the spur invaginating between the two hemicords were seen. Fibrous attachment between the bony spur and the upper lamina was resected []. The bony spur is then resected from its base to expose the opened anterior dura of the spine []. The anterior dura was sutured [], and the spinal roots were demonstrated []. Posterior dura was dissected from both the sides from the subcutaneous tissue and reverted to midline and closure was done in a water tight fashion. Skin defect was large and direct approximation of the lips of the skin was not possible. A wide subcutaneous undermining was done to allow a Z-plasty-type flap closure. However, the post-operative follow-up showed necrosis in the lips of the flap. Sutures were removed in the post-operative day 10. Debridement of the necrotic lips was done and wound was allowed to heal by secondary closure with regular wet dressing and boric acid powder. No neurologic deficits were seen after surgery. |
A 14-year-old girl presented with a history of several weeks of persistent headache, hoarse voice, and 20 lb weight loss with tongue deviation on exam. A brain MRI scan showed a 1 × 2 cm enhancing right-sided skull based mass that was invading the hypoglossal nerve canal (). Biopsy of the mass showed poorly differentiated tumor with round to spindled cells in a myxoid background (). An extensive immunohistochemical panel including positive desmin and myogenin stains was consistent with rhabdomyosarcoma and cytogenetic testing was negative for (2; 13) and (1; 13) translocations, which would be more consistent with alveolar rhabdomyosarcoma. No anaplastic features were noted and the tumor was diagnosed as embryonal rhabdomyosarcoma. The tumor was not amenable to complete surgical resection, and thus the patient was diagnosed with IRS Stage 2, Group III eRMS. The patient received standard chemotherapy with vincristine, actinomycin-D, and cyclophosphamide. Local radiation therapy (RT) was initiated immediately for symptomatic treatment and 50.4 Gy RT to the local tumor produced slow improvement in the vocal cord paralysis and resolution of the tongue deviation.\nApproximately 6 months after beginning chemotherapy and 4 months after the completion of RT, the patient developed lower extremity weakness, gait disturbance, incontinence, and headaches. Imaging revealed new diffuse leptomeningeal metastases involving the entire brain and spine. A ventriculoperitoneal shunt was placed, and, to address symptoms of lower extremity weakness and incontinence, the patient emergently received 30 Gy palliative RT to her lower thoracic spine. Shortly after completing RT, she developed difficulty in breathing, seizures, and altered mental status. With ongoing respiratory failure and neurologic deterioration, the family and medical team decided to transition the patient to comfort care only. The patient died shortly after extubation and permission to perform a complete autopsy was given by the family. The study was conducted with appropriate approval by the Institutional Review Board.\nThe autopsy was performed approximately 28 hours after death. Examination revealed numerous fleshy masses ranging from 1 to 6 cm involving the cerebrum (left frontal and occipital cortex and underlying white matter, cingulate gyrus bilaterally, genu of corpus callosum, left basal ganglia, hypothalamus and left thalamus, right hippocampus, and optic chiasm), cerebellum, and brainstem as well as the leptomeninges. Metastatic tumor was harvested sterilely from several brain sites and placed immediately in RPMI solution with sections fixed in 10% formalin for histology from the same anatomic sites. Histology showed poorly differentiated neoplasm with round to elongated spindled cells in a myxoid background, highly similar to those seen in the previous biopsy (). |
The patient is a 20-year-old female who presented to the emergency department (ED) with the complaint of a diffuse whole-body rash. Her past history was notable for depression as she was recently prescribed lamotrigine two weeks prior to ED presentation. She was recently discharged from another hospital two days ago where she was hospitalized for three days due to complaints of blistering of her lips, sore throat, and mild rash on the extremities. She was given the diagnosis of hand-foot-mouth disease and was treated with 1 gram of ceftriaxone. She reported a decrease in the severity of sore throat upon discharge from ED but the rash continued to progress and had spread throughout most of her body parts. The lamotrigine was subsequently discontinued by the primary care physician. She decided to present to the emergency department (ED) for further evaluation.\nIn the ED, she had a temperature of 102.4 F, blood pressure of 152/67 mmHg, heart rate of 120, and a respiratory rate of 22 per minute with an oxygen saturation of 98%. Physical examination was notable for a young female appearing uncomfortable with a diffuse maculopapular erythematous rash with scattered bullae lesions covering approximately 36% of her body surface area (Figure , ). Her lips were blistered with erythematous mucus membranes as well as erythema with erosions of the eyes (Figure ). The Nikolsky's sign was not appreciated. The rest of the physical examination was unremarkable. The laboratory data were unremarkable for signs of active infection given no leukocytosis and sterile blood cultures. Acetaminophen 1 gram was administered orally for fever and she was resuscitated with intravenous fluids. The patient was admitted to the progressive care unit for closer hemodynamics monitoring and supportive care. She was then transferred to a tertiary level burn care center within the next few hours for the continuation of care. She was diagnosed with toxic epidermal necrosis. She was monitored closely and received supportive management in the burn unit. The patient was subsequently discharged home in one week. |
A 68-year-old female underwent a partial left-sided chest wall resection, with partial removal of the 6th and 7th ribs and of the scapula angle for elastofibroma (). The chest wall defect was reconstructed by using a Mersilene mesh, secured by interrupted pericostal stitches, and covered by a sufficient volume of viable muscles. The postoperative course was uneventful; the radiographic aspect at discharge was normal (). The first symptoms in the form of pains in the region of the incision appeared five months after the operation, and computer tomography (CT) of the thorax showed a lung hernia in the region of the mesh covering the chest wall defect (Figures and ). The patient refused the proposed surgical correction, being only slightly limited in usual daily activities. During the next several months, the symptoms persisted with variable intensity under analgesic therapy, till the moment when pains significantly limited patient's daily activities, 22 months after the operation. The repeated chest CT showed a slight increase in hernia size, with no signs of tumour recurrence (), so that reoperation was planned.\nAfter the excision of the previous skin scar and the incision of the muscular layer, the mesh region was exposed, showing a lung protrusion (4 × 3 cm) along the anterolateral edge of the mesh (). The local situation is schematically presented on . The mesh suture line in the hernia region was completely disrupted, with a small piece of the herniated lung being completely detached from the mesh, the remaining lung surface under the mesh area being fully adherent to the mesh. By careful dissection, the mesh was separated from a firmly adherent lung and removed (). After adhaesiolysis and complete lung liberation, a wedge resection of the afunctional lung tissue of the superior segment of the lingula was done, just in the region of contact with the mesh. After the chest tube insertion, the chest wall defect was reconstructed by suturing a Mersilene mesh in two layers—single pericostal sutures for initial fixation and running suture for additional reinforcement (). A final chest wall stabilization was done by the fixation of two Synthes plates (DePuy Synthes J&J) over the 5th and 6th ribs (). The postoperative course was uneventful. The chest X-ray on discharge, on postoperative day 5, is presented in .\nAt the last contact with the patient, one year after the operation, the general condition was good, without the need for analgesics. |
A 36-year-old patient, gravida 2 para 2, was admitted to the hospital with a complaint of worsening lower abdominal pain occurring on each menses for 8 months. Her external and internal genitalia, including the cervix, were normal except for the 6 cm accessory cavitated left uterine mass suggestive of hematometra that is compressing the urinary bladder without any other genitourinary system pathologies shown on ultrasound examination (). Diagnostic hysteroscopy revealed a single cervix without any vaginal malformations and a relatively small uterine cavity with right tubal ostium and without left tubal ostium. It was decided to remove the left uterine horn (Class U4a/Hemi Uterus)( by the laparoscopic route. Evaluation of the abdominal cavity revealed a left non-communicating rudimentary horn tightly residing on the lateral abdominal wall and two grossly normal ovaries and tubes (). A probable occult occlusion of the tube might be present, and this tubal occlusion might cause this late occurrence. However, neither endometriosis nor any prior tubal or abdominal operation history was noted.\nFirst, to remove the fallopian tubes from the left uterine horn, they were coagulated and divided by careful tissue transection. Second, the vesicouterine peritoneum was divided to create the bladder flap from the cervix and the left uterine horn. Third, dissection of the retroperitoneal space beneath the round ligament to identify the ureter and the left hypogastric artery branches was performed. The broad ligament was fenestrated to lateralize the left ureter and facilitate transection of the utero-ovarian pedicle. The retroperitoneum was dissected, and the ureter tract was followed. Posterior peritoneum was also opened to create distance from the ureter and provide a place for the division of the horn by a monopolar hook. After the dissection and coagulation of the left uterine artery at the origin of the left hypogastric artery to minimize the bleeding during excision of the uterine horn by an advanced bipolar energy device (), the resection of the rudimentary horn was achieved using a monopolar hook(. After controlling the bleeding and irrigating and suctioning the abdominal cavity, no other hemostasis sutures were required, and the operation was completed successfully. The patient was discharged on the first postoperative day, and normal regular menstrual cycles without any pain and complaints during the 6 and 12 months after the surgery were noted.\nThe patient signed an informed consent that allowed us to use her data. |
A 52-year-old man with underlying human immunodeficiency virus (HIV) infection presented with a painful, swollen right knee and fever of two days duration. Examination revealed a tender, warm knee with gross effusion and limited range of motion. Initial aspiration produced 60 mL of pus and confirmed the diagnosis of right knee septic arthritis. The specimen was sent for bacteriologic, tuberculosis and fungal culture and sensitivity test.\nWe planned for emergency arthrotomy washout and debridement of the septic joint but were unable to proceed as he developed acute myocardial infraction upon admission. In view of the acute turn of events which rendered him temporarily unfit for surgical intervention, we performed bedside continuous irrigation and drainage of the septic knee whilst he received medical therapy for his heart condition. Meanwhile, he was empirically started on intravenous cloxacillin one gram six hourly.\nWe used two 14 gauge (orange) cannula; one as inflow inserted to the suprapatellar recess and the other as outflow inserted anterolaterally to the knee joint (). The inflow cannula was connected via a standard drip line to a bag of physiological saline solution (sodium chloride 0.9%) placed on a drip stand at a height of 1.8 metres (). The outflow cannula was attached via a three-way stopcock and drip line to a standard urine drainage bag hung at the side of the bed. We performed once a day intermittent saline distension and drainage for thirty minutes whilst maintaining continuous instillation and drainage by gravity at other times for a total of four days. We monitored for signs of leakage at every shift and kept a meticulous input and output chart.\nThe outflow irrigation fluid became clear after twenty four hours of continuous irrigation. The irrigation system was removed after four days when clinical improvement was seen, as evidenced by absence of pain, resolution of fever and decreased infective parameters (white cell count from 13.4 to 9.7 × 109/L, C-reactive protein level from 47.6 to 6.3 mg/dL and erythrocyte sedimentation rate unchanged 50 to 52 mm/hr). Culture of both blood and knee aspirate revealed Streptococcus pyogenes which was sensitive to penicillin, for which he received intravenous aqueous crystalline penicillin G 2.4 mega units six hourly for six weeks. The patient was advised for a formal washout and debridement when he was more stable but he was reluctant due to high anaesthetic risk. He also did not return for follow up upon discharge. |
A Fifty year old female patient reported to the hospital with complaint of swelling in the lower jaw since 4-5 months. On clinical examination, a swelling was seen below the left side of the angle of the mandible which was measuring 5 × 3cm and extending 4cm below the base of the mandible and obliquely 5cm from the angle of the mandible. The mass was just anterior and deep to the sternocleidomastoid muscle []. On palpation it was soft in consistency, fluctuant and painful. An The FNAC was performed in a municipal hospital which was diagnosed as mucoepidermoid tumor of parotid and was negativity for Koch bacilli. The FNAC report did not mention the detailed findings. Based on these findings the patient was operated on and the excised tissue was grossed. The gross finding showed that the tissue was measuring 5 × 3 cm and cut section was partially brownish in colour. The specimen showed mucoid material within the cystic cavity, the cyst wall was very thick with papillary projections []. The tissue was formalin fixed, processed, sectioned and stained with haematoxylin and eosin.\nHistopathological findings showed cystic cavity lined by epithelium and the underlying connective tissue showed germinal centres. The cystic epithelium was thrown into folds at various places. The epithelium was stratified squamous epithelium type and was 6-8 cells layered thick []. The rete ridges were flat. At some places the epithelium was separated from the underling connective tissue. The cystic lumen showed some squamous epithelial strands which were desquamated from the cystic epithelium. The underlying connective tissue showed abundant lymphoid tissue with many germinal centres. Some fibrous connective tissue elements with fibroblasts and blood vessels lined by endothelial cells were evident. [Figures –]. To rule out tuberculosis Ziehl–Neelsen stain was previously done and there was no evidence of Koch bacilli. Based on all these findings a final diagnosis of infected branchial cyst was given. She underwent complete excision of the lesion with preservation of the facial nerve. The patient recovered well and had no recurrence at 1-year of follow up. |
A 60-year-old post-menopausal female presented to her primary care provider for intractable nausea, diarrhea, vomiting, and unintentional weight loss of 25lb over 2 months. She had a history of poorly controlled type II diabetes mellitus with peripheral neuropathy, and previous cholecystectomy. The patient was adopted, and thus her family history was unknown. Her physical exam was unremarkable. Her primary care provider referred her to a gastroenterologist for an endoscopy due to her clinical picture and a screening mammogram due to never having received one before.\nScreening mammography performed six weeks after her initial encounter showed an oval mass in the upper outer quadrant and focal asymmetry with architectural disturbances in the anterior third of the left breast. Subsequent diagnostic mammography was concerning for breast cancer and biopsies of the mass and suspicious lymph nodes were performed. Her pathology demonstrated the lesion to be grade 2 ER positive (90%), PR positive (2%), HER2/neu positive invasive ductal adenocarcinoma with a Ki67 of 20% (). Due to the size of the oval mass and the presence of cancerous cells on lymph node biopsy, her initial stage was T2N1 stage II triple positive adenocarcinoma of the left breast.\nA week later, an abdominal ultrasound demonstrated a dilated common bile duct of 14mm without obvious stones. Further work-up included an MRI of her abdomen, which demonstrated liver enlargement and a 3.5cm mass in the uncinate process of her pancreas, and diffuse thickening of the stomach wall (). The MRI also demonstrated enhancing vertebral lesions concerning for metastatic disease. Esophagogastroduodenoscopy was performed, visualizing the lesion in the stomach wall (). To determine if the gastrointestinal lesions represented another primary cancer, biopsies of the lesions in the stomach and pancreas were taken. The lesion biopsies were demonstrated to be metastatic breast adenocarcinoma due to immunohistochemical staining positive for estrogen receptors, E-cadherin, and GATA3 ().\nThe patient was ultimately diagnosed as having stage IV triple positive invasive ductal adenocarcinoma of the breast. Once the diagnosis was confirmed, systemic therapy was initiated with docetaxel, pertuzumab, and trastuzumab. |
A 68-year-old female underwent a partial left-sided chest wall resection, with partial removal of the 6th and 7th ribs and of the scapula angle for elastofibroma (). The chest wall defect was reconstructed by using a Mersilene mesh, secured by interrupted pericostal stitches, and covered by a sufficient volume of viable muscles. The postoperative course was uneventful; the radiographic aspect at discharge was normal (). The first symptoms in the form of pains in the region of the incision appeared five months after the operation, and computer tomography (CT) of the thorax showed a lung hernia in the region of the mesh covering the chest wall defect (Figures and ). The patient refused the proposed surgical correction, being only slightly limited in usual daily activities. During the next several months, the symptoms persisted with variable intensity under analgesic therapy, till the moment when pains significantly limited patient's daily activities, 22 months after the operation. The repeated chest CT showed a slight increase in hernia size, with no signs of tumour recurrence (), so that reoperation was planned.\nAfter the excision of the previous skin scar and the incision of the muscular layer, the mesh region was exposed, showing a lung protrusion (4 × 3 cm) along the anterolateral edge of the mesh (). The local situation is schematically presented on . The mesh suture line in the hernia region was completely disrupted, with a small piece of the herniated lung being completely detached from the mesh, the remaining lung surface under the mesh area being fully adherent to the mesh. By careful dissection, the mesh was separated from a firmly adherent lung and removed (). After adhaesiolysis and complete lung liberation, a wedge resection of the afunctional lung tissue of the superior segment of the lingula was done, just in the region of contact with the mesh. After the chest tube insertion, the chest wall defect was reconstructed by suturing a Mersilene mesh in two layers—single pericostal sutures for initial fixation and running suture for additional reinforcement (). A final chest wall stabilization was done by the fixation of two Synthes plates (DePuy Synthes J&J) over the 5th and 6th ribs (). The postoperative course was uneventful. The chest X-ray on discharge, on postoperative day 5, is presented in .\nAt the last contact with the patient, one year after the operation, the general condition was good, without the need for analgesics. |
A 22-year-old male patient was admitted in the surgery ward with symptoms of bowel obstruction. He had a history of vomiting and abdominal distention since 10 days. The patient had experienced similar complaints on and off for the past year. On examination, the patient's abdomen was distended and a vague swelling was noted in the umbilical region. The baseline investigations were normal with only a rise noted in the patient's erythrocyte sedimentation rate which was 70 mm at 30 minutes and 118 mm at the end of 1hour. His Mantoux tested negative. An X-ray abdomen showed multiple fluid levels and a contrast-enhanced CT scan revealed ileal wall thickening and mesenteric nodes. The surgeon proceeded with a laparotomy and resected a segment of ileum with omental adhesions and sent for histopathological examination.\nThe cut open small intestinal segment measured 34 × 6 cm. A polyp of 1.5 cm diameter was noted 18 cm from the proximal end. Two ulcers were identified, the first measuring 2 × 2.5 cm was 14 cm from the proximal end and the second, measuring 2 × 1.5 cm was 20cm from the proximal end. Both ulcers were shallow, situated along the horizontal plane, with undermined edges and a fibrotic base. A stricture was present 9cm from the distal end with dilatation of the proximal segment. The serosa showed large lymph nodes measuring 4 × 5 cm, whose cut section was whitish and creamy in consistency. The areas adjacent to the ulcers showed diffuse thickening of the wall and on cut section revealed a whitish mass extending from the submucosa upto the serosa. Representative sections from each of the grossly abnormal areas and the resected margins were submitted for processing.\nSections from the polyp showed a polypoidal mass with a core containing smooth muscle fibers and was identified as a Peutz-Jeghers polyp. Sections from the ulcerated areas showed edematous lamina propria with a dense infiltration of eosinophils and lymphocytes with congested blood vessels in the submucosa and a dense lymphocytic infiltration in the muscularis propria [] and serosa. Sections from the lymph node showed partial loss of architecture, areas of necrosis and fibrosis with epithelioid cell aggregates but no definite granuloma was made out. Multiple sections from the thickened areas showed diffuse sheets of lymphocytes ulcerating the mucosa and infiltrating transmurally upto the serosa and periserosal fat. Rest of the mucosa showed focal erosions, increased eosinophilic and lymphocytic infiltrates in the lamina propria and increased intra-epithelial lymphocytes with focal shortening of villi. In view of the high suspicion of tuberculosis and lymphoma, further diagnostic modalities were sought after such as acid fast staining, polymerase chain reaction (PCR) and immunohistochemistry.\nZiehl–Neilson acid fast staining revealed numerous acid fast bacilli. The PCR was positive for tubercle bacilli. A lymphoma panel of immunohistochemical markers showed CD 45 and CD 20 positivity [] with weak positivity for Bcl2. The rest of the IHC markers such as cytokeratin, chromogranin, CD 3 and CD 10 were negative.\nThus, a final diagnosis of primary small intestinal B-cell lymphoma, tuberculosis and Peutz-Jeghers polyp was made. |
I am a 35-year-old Chinese female family physician in Hong Kong and would like to share with you my story. I tore my left anterior cruciate ligament while playing hockey and had reconstruction surgery done more than ten years ago. The surgery was uneventful but unfortunately my knee became unstable again one year later which I attributed to poor rehabilitation on my part. Despite this, I continued with regular swimming practice although I never played hockey again. I developed an interest in hiking a few years ago and during the winter months I would go hiking every week for three to six hours. The instability in the left knee was not troublesome as long as I remained careful. Last year I climbed to the top of Mount Kinabalu (4095 m) and had no problem with my knees at all.\nThis year to challenge myself, I took part in a 50 km charity hiking event. It was the longest hike I had ever done, having completed the whole trail uneventfully within twelve hours. My knees felt fine until the following evening when the right knee, the 'good' one, started to hurt, particularly around the patella and yet there was no problem with my operated knee. There was no effusion and the range of movement was full. I thought it was patellar tendonitis, so I took diclofenic sodium 75 mg daily for five days. Yet the pain persisted and even got worse. I switched to acetaminophen 1 gm four times daily for two days, but again it did not help. The pain occurred not only when I walked on level ground but also while I was at rest. It even woke me up on a few nights. Eventually I visited a physiotherapist, twelve days after the hike. She examined my knee and it hurt when she compressed directly on my patella. I also had severe pain when she asked me to squat on my right leg. Her impression was patellofemoral syndrome and suggested acupuncture. Since the pain was absolutely terrible and it did not improve with medication, I decided to give it a try.\nI had acupuncture treatment on three consecutive days and three times weekly thereafter. Ultrasound therapy was applied for five minutes prior to acupuncture. Seven needles were inserted around the knee (Figure ) for thirty minutes and during which the physiotherapist would twirl the needles every ten minutes. Hot pack was then applied at the end for ten minutes. I was also instructed to do a series of stretching and muscle strengthening exercises at home every day for an hour. The progress was slow after six sessions, so patellar taping was applied. Unfortunately I developed a severe allergic reaction to the tape with pronounced redness, swelling and itch at the end of the day. The tape was removed and I continued the ultrasound therapy, acupuncture and had ice pack instead of hot pack. The pain and the rash gradually improved and by the fourth week of physiotherapy, I was able to resume full activity and my usual swimming practice with no pain at all. I had twelve acupuncture sessions altogether and the pain was completely gone within six weeks.\nI reviewed the literature and found that patellofemoral syndrome is indeed quite a common condition although consensus has not yet been reached in regard to its aetiology and treatment []-[]. Jensen et al. suggested that acupuncture may be an alternative treatment for patellofemoral syndrome []. Various reviews have tried to examine the efficacy of different forms of treatment, including exercise therapy, acupuncture, patellar taping, use of orthotics and braces and pharmacotherapy []-[]. However further high quality, prospective, randomized, controlled, long term studies with validated outcome measures are still needed to develop treatment models for patellofemoral syndrome.\nI must have overused and overloaded my right knee to compensate for the instability in my left knee although I was unlikely to be aware of that. I am not sure which modality helped me most, whether it was the ultrasound therapy, acupuncture, the hot or cold pack, the stretching exercise or the muscle strengthening exercise, but I am glad the pain has gone for two months now and I hope it will not recur. |
A 59-year-old woman had noted moderate pruritus in the upper area of her left breast and sensed annoying mass in the same area, 15 months before she was referred to Al Zahra hospital where we took her history notes. One month after the initiation of pruritus, bilateral diagnostic mammography (4 films) showed a 1 cm spherical mass with spiculated edge in the upper zone of the left breast suspected to a malignant lesion and suggested fine-needle aspiration cytology for further studies according to breast imaging-reporting and data system (BIRAD V). Breast sonography revealed normal skin, nipple and subcutaneous fat but a 8.2 × 7.2 mm irregular contour, hypo echo lesion, 1 cm from nipple in the position of eleven o’ clock. Solid mass did not existed in the right breast. Due to these findings the patient underwent partial mastectomy and left axillary dissection 2 month after onset of mass sensation. Post operative pathobiopsy showed an invasive ductal carcinoma. Pruritus subsided immediately after surgery. Lung metastasis and post surgical collection were reported in multi 64-slice CT scan with contrast, 20 days after operation. Lungs were free from any pathologic infiltration. There were multiple pulmonary nodules (one 7 mm in the right upper lobe and three tiny foci in the left lung with mean size of 4 mm) in lung fields. No mediastinal or hilar lymphadenopathy, plural effusion and thickening were seen.\nDuring 6 sessions of chemotherapy and 31 sessions of radiotherapy, the patient had no considerable symptom or problem until 1 week after last session of radiotherapy when she presented with severe scattering pain of the whole left upper limb with a radiation to scapula. Multiform blisters appeared 1 week after, when the scattering pain was decreasing. The watery/vesicular blisters were in the distal flexor surface of the arm in different sizes, dominantly in the cubital area lasted for 3 weeks. These clinical manifestations were diagnosed as herpes zoster activation and approved by positive direct fluorescent antibody test for herpes zoster. The patient was hospitalized and intravenous acyclovir was initiated 10 mg/8 hours for 7 days. Gradual improvement of blisters and severe pain was observed during treatment. A few days after completion of treatment, itching and motor involvement including elbow flexion and shoulder abduction appeared suddenly. The range of motion of glenohumeral joint decreased but the patient was able to adduct shoulder, flex and extend wrist and passively extend elbow. Magnetic resonance imaging showed compression of the left nerve root at C4-C5 and C5-C6 levels by protruded disk. No evidence of metastatic brachial plexus was noted []. Acromioclavicular and glenohumeral joint osteoarthritis were found in the left shoulder CT scan. No fracture or dislocation was reported.\nSensory nerve conduction study and electromyography revealed low amplitude action potentials in left superior radial and median (from thumb and index) SNAPs (sensory neuron action potentials) and nascent MUAPs (motor unit action potentials) in left deltoid muscle [Tables and ]; also, a low amplitude action potential in left radial nerve was seen. Above findings were compatible with zoster-induced radiculoplexitis considering the MRI results, patient's history and clinic.\nWe examined the patient 5 months after beginning of pain and rash when 34 sessions of physiotherapy were performed to recover motor activities. The patient's neuropathy and neuroplexitis palliated gradually but pain and itching made poor decrement, suggesting post herpetic neuritis (PHN), as we find in immunocompromised cases.[] She was able to flex elbow actively and abduct shoulder about 30 degrees while she could not do it before.\nAt the present time, rehabilitation is supposed to be continued, focusing on biceps brachii and deltoid muscle reinforcement and increasing left upper limb range of motion under supervision of orthopedic physician. |
A 43-year-old man with a history of two failed endocardial ablation procedures at the right ventricular outflow tract (RVOT) for recurrent, drug-resistant ventricular tachycardia (VT) was referred to our institution for repeat electrophysiologic study and catheter ablation. Twelve-lead ECG during sinus rhythm showed no remarkable abnormalities (). ECG recorded during VT showed inferior axis and left bundle branch block type morphology, which suggested an RVOT origin (). Transthoracic echocardiography showed mild dilatation of the left ventricle (diastolic diameter 58 mm) with normal left ventricular systolic function (ejection fraction 69%). Gadolinium-enhanced cardiac magnetic resonance imaging before ablation showed no evidence of scar. The patient did not have a past medical history of syncope or any family history of sudden cardiac death.\nDuring electrophysiologic study, clinical VT was spontaneously induced, sustained for minutes, and recurred invariably with isoproterenol infusion. RF energy was delivered repeatedly at the posterior RVOT (), where the earliest ventricular activation of bipolar electrogram preceding the onset of QRS by 30 ms and a QS pattern of unipolar electrogram were recorded during VT (). Ablation using a quadripolar catheter with a 3.5-mm irrigated distal electrode and power control mode at a setting of 30–45 W resulted in the first episode of VT termination within 30 seconds of starting the sixth RF application, but VT recurred as soon as the RF energy was turned off. In total, ablation was attempted 13 times at the RVOT, but sustained VT remained. Epicardial access was obtained percutaneously via the subxiphoid area, and mapping of the epicardial area adjacent to the endocardial RVOT revealed bipolar activation preceding the onset of QRS by only 10 ms, with an rS pattern on unipolar recording. Epicardial ablation was not attempted because the VT was suspected of having an intramural origin within the posterior wall of the RVOT since a long RF ablation time was required to eliminate the tachycardia. Subsequently, the RCC was mapped after identification of the right coronary artery (RCA) ostium and confirmation of the absence of organic stenosis by coronary angiography (CAG; ). Although an initial positive and upright electrogram was not observed in the bipolar recording made at the RCC, an early activation and a QS pattern were recorded in the bipolar and unipolar recordings, respectively, during VT (). Excellent pace-mapping was also obtained at that site. shows the relative positions of the RCC ablation site (white stars) and the RCA. The ablation site was seen to be sufficiently anterior to the RCA in the right anterior oblique view. In addition, the RCA ostium (, white arrows) was seen to be much higher than the ablation catheter tip. Based on these considerations, a decision was made to attempt ablation at this site (). RF energy was delivered using a quadripolar catheter with 4-mm nonirrigated distal electrode, using a temperature control mode with a setting of 50°C and maximum power output of 35 W. During the second RF application at the site ( and ), VT was terminated transiently but recurred after RF energy was turned off. A subsequent third and final RF application at the same site resulted in VT termination in 23 seconds, and the remaining repetitive ventricular beats eventually were eliminated 80 seconds later. In summary, RF applications were applied at the RCC 3 times for a total of 195 seconds, with a maximum impedance drop of 24 Ω (from 117 to 93 Ω), which was not accompanied by any impedance rise. One minute after isoproterenol infusion at 1 µg/min was started after RF, the patient complained of chest discomfort. ECG showed ST-segment elevation in the inferior leads (). Urgent CAG revealed acute severe stenosis at the ostial RCA, which was not resolved by repeated intracoronary nitroglycerin injection (, top). Intravascular ultrasound examination showed eccentric intimal thickening (, top, white arrow), which resolved after angioplasty (, bottom). Both coronary artery dissection and thrombus occlusion were ruled out. Stenting was deferred at that time because subsequent percutaneous balloon angioplasty successfully improved the severity of stenosis (, bottom). During 18-month follow-up, CAG repeated 3 times after the ablation procedure showed gradual progression of stenosis in the ostial RCA, which eventually required stenting. The patient continued taking beta-blockers and calcium channel blockers. Fortunately for the patient, the burden of VT episodes greatly improved from almost daily to a few times per year. |
A 46-year-old man also reported exercise/activity-related pain and fatigue from early childhood. As patient 1, he was always considered to be “a lazy child”. He was not able to run and physical activities such as walking or swimming were challenging. As a child he reported trying to build a good relationship with his physical education teachers, so they would feel pity for him and he could avoid any strenuous physical activities (PA). He experienced three severe episodes of rhabdomyolyses in his life. The first one happened during childhood, which followed vigorous physical activities. The second rhabdomyolysis episode was at the age of 18 following multiple squats, which he performed during a physical test for military service. Not qualifying for the military service, instead he had to complete a civilian service at the university hospital at the age of 22. As he was clearly weaker than his colleagues, a diagnostic investigation took place, which included a skeletal muscle biopsy. At that time, doctors advised him to avoid excessive physical effort. He was told about the second wind phenomenon; however, it was not explained how to reach it. Additionally, he was recommended to eat a maximum of 20% of his daily food intake in carbohydrates. However, the rest of the macronutrients recommendations were not specified. In the following years, his physical activity level decreased, leading to physical deconditioning, loss of muscle mass, and increase in fatty adipose tissue and body weight.\nThe third rhabdomyolysis episode was experienced one year ago and was nearly fatal. He was undergoing physical assessment for the insurance company and pushed himself too hard, resulting in a severe contracture of his lower back muscles. He was admitted to hospital. During the hospital stay he was also diagnosed with myocarditis. Following this episode, he decided to learn more about the condition to manage it better.\nHe began doing gentle aerobic exercises, which improved his ability to attain a second wind. He attended a conference where he met a Spanish team from whom he learnt about the strength training trial performed in Madrid and decided to try it himself in a local gym []. Initially he approached a personal trainer. However, he did not feel the trainer understood his condition and was pushing him too hard. He decided to write his own programme based on strength training principles and exercises he learnt at the conferences and meetings.\nWhen in the gym he reports always doing an aerobic “warm up” by cycling on a stationary bike for 20 minutes, he then exercises on resistance machines (chest press, seated row, butterfly, chest horizontal adduction, reversed butterfly, rare deltoids and trapezius, lat pulldown, leg press, leg adductors, and leg abductors). He finishes his sessions with more aerobic exercise by walking on a treadmill and/or cycling on a stationary bike. and illustrate his progress during the first three months of training (based on personal records). He completes between 5 and 15 repetitions of each exercise with one-minute rest in between sets. He stops a set of exercise earlier if he feels any discomfort in the muscle.\nHe did not report Delayed Onset Muscle Soreness (DOMS) or McArdle symptoms following initial sessions. After three months of resistance training, he found his sleep pattern, overall stamina and McArdle symptoms improved. He is now able to walk two kilometres stopping only once, as opposed to previously when he would have to stop multiple times. He has also found it easier to perform everyday tasks such as changing a car tyre. He has been eating a balanced diet. He autonomously chose not to take any supplements containing glucose pre- or intratraining session. |
A 28-year-old male laborer who is right hand dominant and works with his hands for a living with a body mass index of 45 kg/m2 presented to the emergency department (ED) with swelling and pain at the distal aspect of his left thumb with no known injury. The patient reported that he had been seen by a provider over 4 weeks previously and was treated for cellulitis of the hand with antibiotics. At that time, no radiographs were taken. During the 2 weeks before presenting to the ED, the patient stated that the swelling had gradually increased and that his pain level had increased to 8/10. Radiographs were performed in the ED and the hand surgeon was subsequently consulted (). The radiographs displayed a lytic, disruptive, and mildly expansile lesion of the distal phalanx of the first finger which concerned the consulting physician for sarcoma (). The patient was started on Ibuprofen and Tylenol #4 and seen in the office for evaluation. At this time, it was found that the patient had redness, swelling, and weakness performing left thumb pinch. The patient denied any history of trauma, fever, bruising, decreased range of motion, numbness, tingling, and purulent drainage. His quick dash score at this time was 39. Repeat radiographs were performed and again displayed diffuse bone formation of the distal phalanx of the left thumb concerning for a destructive osteolytic tumor. The option of obtaining an MRI for surgical planning and further assessment of the tumor was discussed with the patient, however given the radiographical findings, the patients uninsured status and his desire for a surgery that would allow him to return to work as quickly as possible, an MRI was not obtained. After discussing treatment options and further workup, an excisional biopsy of the lesion was planned.\nTwo days later, the patient underwent an excisional biopsy of a 22 mm × 22 mm tumor of the left thumb with a 1–2 mm subcutaneous tissue margin and bone sampling. Pre-operative photographs demonstrate the left thumb mass with erythema and nail deformity (). Intraoperatively, an incision was made along the glabrous versus non-glabrous margin of the distal phalanx along the axis of the left thumb to remain in the appropriate orientation of the incisions. This was done in case the patient required further amputation or resection. The dissection was then carried into the subcutaneous tissue with a fish-mouth design of opening. On approach to the periosteal tissue, it was found that the patient had a large tumor resting within the site that should normally be occupied by the shaft of the distal phalanx. There was complete loss of the cortical surface with a well-contained mass that did not seem to invade into the subcutaneous tissue. The identified tendinous tissue, nail bed tissue, and remaining bone appeared to be otherwise healthy. Using a bone cutter, a portion of the bone was excised out and the skin margin was carefully dissected making an excisional biopsy of the mass. The curette was then taken to carefully clear and scrape questionable appearing tissue which was gathered and sent to pathology. The wound was then thoroughly irrigated, and the fish-mouth flaps were loosely re-approximated with interrupted nylon suture.\nThe pathology demonstrated features of GCT of the bone including numerous osteoclastic giant cells associated with polygonal stromal cells with areas of spindle cell differentiation and hemorrhage (). The stromal cells showed scattered mitotic figures and mild nuclear atypia; however, they showed no evidence of malignant cytology (). In addition, there were areas of reactive bone formation found within the tumor (). This was confirmed by a tertiary care center. The patient was then evaluated 10 days later in the office and his quick dash score at this time was 52. He was informed of the pathology results and the treatment options available to him. Given the patients desire to return to work as quickly as possible and the fact that the tumor was in his non-dominant hand, he elected to undergo left thumb distal phalanx amputation without reconstruction. This would allow him the fastest recovery time and shortest return to work. The risks and benefits of surgery as well as surgical treatment, including reconstructive options, were discussed extensively with the patient. The amputation was completed after examining the mass with the necrotic tissue of the distal pulp and nail bed being excised (). In addition, the eponychial fold, paronychial fold, and other nail forming structures were excised (). The remaining distal phalanx was examined and determined to be composed of only the articular surface and some remaining attachment of the flexor pollicis longus and extensor pollicis longus tendons. This was debrided and cleaned and then the volar skin was molded to fit the dorsal rim of tissue in a trapdoor fashion. The excised portion of the left thumb was sent to pathology and it was determined that the margin of excision was viable (). The sample showed one focus of epidermal inclusion cysts with secondary acute inflammation and fibrosis which was determined to be benign.\nThe patient had an uneventful post-operative course and continues to do well at 1-year post-surgery with radiographs demonstrating a well healed left thumb (, ). He has had a normal return to work with complete adaptation to the amputation. |
A 66-year-old right-handed gentleman was referred for treatment of a giant cell tumour of the right distal radius. He had suffered with gradually worsening wrist pain and swelling for approximately six months without any previous history of trauma. Symptoms were worse at night and function was poor. He was otherwise fit and well with no other complaints. He had a colonic tumour resected eleven years ago and was clear of disease at his last check up. On examination, a firm, diffuse swelling was present on the dorsal aspect of the distal forearm and all movements of the right wrist were reduced secondary to pain. Wrist flexion and extension were limited to 10° each while radial deviation was absent and ulna deviation was reduced to 10°. Pronation and supination were reduced to 30° and 20°, respectively, from neutral. In comparison to the contralateral side, clinically, grip strength of the affected limb was significantly decreased. No neurovascular deficit was present. Radiographs of the forearm and wrist demonstrated a large lytic lesion of the distal radius, suggestive of a giant cell tumour of bone (). This was confirmed following biopsy and a chest radiograph, whole body bone scan and MRI of the forearm revealed no other lesion.\nIt was decided that, in view of the size of the lesion and the patient's age, resection of the tumour and subsequent reconstruction of the defect were to be performed using a distal radial endoprosthesis which articulated with the carpus. A custom-made predominantly titanium implant (Stanmore Implants Worldwide Ltd.) was constructed for use, which included a fixed cobalt-chrome articulating surface. Measurements for accurate manufacture of the implant were taken from radiographs of the contralateral forearm (). It was essential that an anatomical fit was achieved to enable maximum function of the flexor and extensor tendons about the wrist joint. The implant included a tight-fitting proximal intramedullary stem and a hydroxyapatite(HA)-coated collar at the site of the bone-prosthesis interface to aid osseointegration. The requirement and design for this HA collar were based on previous the literature demonstrating both the growth of bone into and around the HA material, enhancing stability, as well as a decreased rate of aseptic loosening with massive endoprostheses [, ].\nVia a dorsal incision, 70 mm of the distal radius, including the lesion, was excised along with the surrounding periosteum and the biopsy tract. It was confirmed that the cortex around the lesion was completely intact. The proximal radius was reamed and the prosthesis was inserted using gentamicin bone cement. Adequate reduction was ensured to provide maximum contact between the proximal hydroxyapatite collar and the distal cortex of the freshly cut bone. The stability of the wrist was then enhanced using the extensor carpi radialis longus, which was transected distally, passed through a premade design hole in the lateral section of the distal prosthesis and reattached to the capsule to form a lateral stabiliser. The capsule was then sutured down onto the soft tissue around the distal ulna, haemostasis was achieved and the incision was closed in layers. The limb was placed into an above-elbow plaster of Paris splint for four weeks. Post-operative radiographs suggested that the distal radius prosthesis was longer than intended despite the prosthesis sitting perfectly on the bone at the level of the resected radius.\nAt four weeks post-operatively the plaster of Paris splint was exchanged for a future splint. At six weeks, hand and wrist physiotherapy was initiated to start wrist flexion, extension, and rotation. The patient was followed up post-operatively with regular clinical and radiographic evaluation to assess symptom relief, hand and wrist function, and implant survival. At the most recent review, at 56 months, he achieved wrist dorsiflexion of 40°, palmar flexion of 20°, radial deviation of 10°, and ulna deviation of 20°. Elbow flexion, extension, and pronation were all full while supination reached 45°. On clinical assessment, the wrist was stable and the power of grip in the operated hand was full, equal to that of its contralateral counterpart. Hand and wrist function had improved significantly following surgery to give the patient satisfactory pain-free movement, allowing him to adequately carry out routine daily activities without difficulty. The patients functional score using the full DASH (Disabilities of the Arm, Shoulder and Hand) scoring system was 10.3 out of 100 []. A preoperative DASH score was unavailable for comparison but the intention of this study was not to make comparisons with preoperative function but to assess the outcome of the prosthesis with regards to function and survivorship.\nThe most recent radiographs demonstrated a secure fixation of the prosthesis without any signs of loosening around the intramedullary stem or recurrence of disease. The X-rays confirmed bone remodelling and osseointegration at the bone-prosthesis interface promoted by the proximal hydroxyapatite-coated collar. Due to the position of the distal articular surface of the prosthesis in comparison to the more proximal ulna styloid, there was some ulna translation of the carpus but this had only produced mild degenerative changes of the carpal bones, present along with age-related osteopenia (). |
The patient is a 35-year-old lady presented to the primary care physician with the chief complaint of cough. Her past medical history is significant for a possible tuberculosis infection 15 years ago. She was in direct contact with another patient who was positive for tuberculosis at a gathering event and later completed a six-month course of anti-tuberculosis regimen prescribed by her primary care physician. She otherwise remained to be asymptomatic and healthy. Her nonproductive cough began three weeks ago. She denied fever, chest pain, dyspnea, night sweats, and weight loss but did complain of sore throat and occasional chills. She lived at home with her family and reported to have sick contact with her kids as they were suffering from an upper respiratory infection. She decided to present to the office for further evaluation due to concerns about possible tuberculosis reactivation.\nHer initial vital signs on presentation were as follows: blood pressure 116/59 mmHg, heart rate 81 beats per minute, temperature 97.8°F, body mass index 20.97. Physical examination was completely benign. There was a low clinical suspicion of active tuberculosis given her overall clinical impression. She was recommended to follow up in the clinic in a few weeks after obtaining a two-view chest x-ray (CXR), sputum acid-fast bacillus (AFB) smear, and culture. The patient was followed up via telemedicine over the next few weeks. She continued to have a mild cough with a low clinical suspicion for tuberculosis. The AFB smear and culture could not have been completed due to a lack of sputum samples. CXR revealed small rim calcified lesions in the right mid and upper lung zones suggestive of a benign etiology without findings of active or latent tuberculosis infection (Figure ). The patient was recommended to schedule a follow-up appointment in six months while maintaining a close observation of her symptoms.\nSix months later, her cough reoccurred after an initial improvement over the past few months. The cough was accompanied by occasional sputum. She denied fever, chills, night sweat, sore throat, chest pain, dyspnea, or weight loss. She had been in close contact with people who were diagnosed with an upper respiratory infection. She was recommended to undergo a pulmonary function test (PFT) as well as a chest CT for further evaluation. She was prescribed dextromethorphan for symptomatic relief, educated on the symptoms of active tuberculosis, and instructed to give frequent updates of her symptoms. CT with intravenous contrast revealed a high right paratracheal and medial right apical heterogeneous soft tissue with central areas of calcification (Figure ). She agreed to opt for a routine endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA) to rule out malignancy.\nBronchoscopy inspection revealed unremarkable trachea, left upper lobe, left lingula, left lower lobe, right middle lobe, and right lower lobe; but interestingly, no right upper lobe was observed. Furthermore, EBUS-TBNA showed a right paratracheal mass without surrounding lymphadenopathies that was biopsied and sent to pathology for analysis. The presumptive diagnosis for the right paratracheal mass was likely due to a nondeveloped right upper lung with associated scarring and atelectasis. The pathology report later confirmed the finding as there was no indication of malignancy or fungal infection. |
A 27-year-old female patient has been reported to the department of oral medicine and radiology with the chief complaint of swelling in the lower right front teeth region of the jaw for 2 years. The patient's history revealed that initially the swelling was smaller in size and has increased gradually to the present size. The swelling was not associated with pain or any other symptom. The patient's past dental history revealed the extraction of left maxillary posterior tooth 8 months back due to caries and the healing was uneventful. On extraoral examination, single localized swelling with diffuse margins of size approximately 2 cm × 2 cm was present on lower one-third of the right side of the face below the corner of the mouth with normal-appearing overlying skin [Figure and ]. On palpation, the swelling was afebrile, nontender and firm to hard in consistency. On intraoral examination, single diffuse swelling of size approximately 1 cm × 2 cm was present on the lower right buccal vestibule in relation with 43 and 44 regions, obliterating the buccal vestibule []. The swelling was nontender and hard in consistency. Right permanent maxillary first molar was missing, left mandibular canine and second deciduous molar were over retained and left mandibular permanent canine and second premolar were clinically missing. On the basis of history and clinical examination, a provisional diagnosis of central ossifying fibroma was given. Orthopantomograph (OPG) was advised which revealed multiple bilateral radiopaque sclerotic lesions surrounded by peripheral radiolucent rim apical to majority of the mandibular teeth, with no evidence of root resorption and impacted left mandibular permanent canine and second premolar []. Computed tomography (CT) scan revealed the presence of an expansile lytic lesion in the right side body of the mandible measuring 3.5 cm × 2.9 cm with hyperdense matrix and intact cortex and a small lesion on the left side body of the mandible with a connection in between [Figure and ]. The biochemical investigations were advised which showed increased alkaline phosphatase levels. Thus, a working diagnosis of FCOD was given. Incisional biopsy was taken from the lower right buccal vestibule region and was sent for histopathological examination. The histopathologic section showed numerous woven bony trabeculae in a fibrocellular stroma with numerous ossicles and few cementicle-like areas. Stromal cells were spindle-shaped and arranged in haphazard manner. Osteocytes and rimming osteoblasts were evident []. The histopathological picture was suggestive of cemento-osseous dysplasia (COD). Thus, the final diagnosis of FCOD was confirmed. The treatment done was facial recontouring under local anesthesia, and the patient is on regular follow-up with no signs of recurrence []. |
We present a 42-year-old female with a history of hypothalamic glioma and resultant obstructive hydrocephalus that had been subjected to multiple surgeries since her childhood due to CSF shunt malfunction. Following her last VPS implantation, symptoms of high intracranial pressure (ICP) notably improved but ventricle enlargement remained almost unchanged on computed tomography (CT) [-]. In addition, a coiled distal catheter was found on the abdominal X-ray, which was misinterpreted, probably due to patient obesity, as being in the correct position inside the peritoneal cavity [].\nIn the present admission, 2 months after the latest VPS placement, the patient complained of severe headache, vomiting, and gait disturbance. An urgent cranial CT demonstrated hydrocephalus in addition to a large low-density subdural collection over the right hemisphere with a minor right interhemispheric component [-]. Macroscopic integrity of the whole shunt system was checked on plain radiographs, but because of a suspected dislocation of the peritoneal tip, a CT scan of the abdomen was performed. This revealed multiple loops of the distal catheter and CSF fluid accumulation at the subcutaneous space []. Valve reservoir pumping demonstrated rapid reexpansion and the CSF was easily tapped. CSF Gram stain and culture revealed no microorganisms. Blood count and chemistries were also normal.\nThe paradox of finding a subdural accumulation (typically associated with VPS overdrainage) and hydrocephalus in a patient with distal VPS malposition generated perplexity and uncertainty for us regarding the most appropriate surgical strategy. Draining the subdural accumulation was considered in addition to repositioning the peritoneal catheter. Nevertheless, the delayed development of the subdural collection following several weeks of hydrocephalus in a patient with shunt malfunction led us to consider that both abnormal fluid accumulations might be related to the blocked CSF flow. Thus, we finally recommended VPS revision alone. We first performed a minilaparotomy, which revealed a pseudocyst encompassing the distal end of the catheter. Once adequate CSF outflow was verified, the fibrous capsule was partially resected, and the catheter repositioned intraperitoneally. Following surgery, all symptoms resolved, and postoperative CT performed on day 4 showed resolution of both hydrocephalus and subdural CSF accumulation [-]. Moreover, postoperative abdominal X-ray confirmed correct placement of the distal peritoneal catheter []. After the 2-month follow-up, the clinical and radiological courses remain satisfactory [].\nWe wish to highlight both the time frame of the development and resolution of the subdural accumulation, as well as the successful surgical strategy chosen. CSF accumulation occurred in a delayed fashion following several weeks of ventriculomegaly, and it was completely resolved by the 4th postoperative day after only VPS revision, which points to a shift of excessive CSF from the ventricles to the subdural compartment as the underlying mechanism for subdural fluid accumulation in our patient. |
A 19-year-old female patient presented to the department with a chief complaint of discoloured anterior teeth. The patient had completed her orthodontic treatment about 1 year back and had also undergone an orthognathic surgical procedure. On clinical examination, teeth # 7, 8, 9 and 10 displayed a yellowish brown discoloration. Grade I mobility was evident in teeth # 7 and 10 and grade II mobility in teeth 8 and 9. On percussion teeth #8 and 9 gave a typical metallic sound as compared to adjacent teeth. An unsuccessful attempt at access cavity preparation was detected on the palatal aspect of tooth #9. Teeth #8 and 9 failed to respond to thermal and electric pulp testing while teeth #7 and 10 responded within normal limits. A periapical radiograph revealed severe replacement of root structure of teeth #8 and 9 with alveolar bone with only half the root lengths persisting []. Some amount of root replacement was also seen in teeth #7 and 10. No canal spaces were evident in teeth #8 and 9; hence, no attempt was made to initiate any treatment. It was planned to take periodic periapical radiographs to assess the progression of replacement resorption in teeth #7 and 10. The patient was explained the poor prognosis but at the same time was reassured that there was no exigency in extracting the teeth. Composite laminate restorations were done for teeth #7, 8, 9 and 10 to mask the discoloration. A follow-up after 1 year revealed no significant increase in teeth mobility and resorption rate.\nExternal apical root resorption is commonly associated with orthodontic therapy due to certain biological factors (genetic susceptibility, systemic diseases associated with phosphorous-calcium metabolic alterations, medication intake) and mechanical factors (tooth movement type, orthodontic force magnitude, duration and type of force).[] Hence, it is essential to take periodic periapical radiographs during treatment.[] When root resorption is detected during active treatment, a decision must be made as to whether to continue, modify or discontinue the treatment.[] Luxation injuries are reported as the principal cause of replacement resorption.[] In this case orthodontic therapy was probably the etiologic agent which also initiated calcific metamorphosis in teeth #8 and 9. Replacement resorption is a continuous process whereby the tooth is gradually replaced by bone.[] Teeth undergoing this form of resorption, have a characteristic metallic percussion sound[] as was observed in this case. There is no known treatment for this type of resorption[] though unsuccessful attempts at Ca(OH)2therapy to arrest the resorption have been tried.[] If the teeth are in a satisfactory position, there is no urgency for tooth replacement because the replacement usually progresses at a slow rate ranging from few months to years. This provides time for the patient to be mentally and financially prepared for the treatment planned by the clinician.[] |
A 63-year-old woman was admitted to our department because of pain and limitation in motion in the left hip for 2 years after primary total hip arthroplasty with hybrid prosthesis for avascular necrosis of the femoral head 15 years ago. The patient underwent mitral commissurotomy because of mitral stenosis 30 years ago, when she complicated with atrial fibrillation, since then she had been taking aspirin 100 mg once daily. The patient presented absolute arrhythmia and uneven first heart sound on auscultation. Electrocardiogram demonstrated atrial fibrillation. X-ray film of the pelvis and the left femur revealed osteolysis around the femoral stem and subsidence of femoral component.\nThe patient was diagnosed as aseptic loosening of the femoral component after total hip arthroplasty and underwent revision total hip arthroplasty with cementless acetabular component and cementless modular stem and allograft for bone defects in both sides. Aspirin was discontinued 7 days before surgery. The procedure was performed uneventfully. The patient began to take rivaroxaban 10 mg 12 h after surgery, which was planned once daily for 35 days. The patient was told to start functional exercises of the left lower extremity as soon as tolerated and began to ambulate with crutches with the left lower extremity free of weight bearing on the 15th day postoperatively.\nThe patient felt sudden numbness, tingling in the right lower extremity, and inability to move it after having a nap on the 17th postoperative day. Physical examination: pulse 80/min and blood pressure (BP) 120/70 mmHg mentally conscious, with pupils round, symmetrical, and sensitive to light. There was loss of sensation, paresis, and decreased skin temperature for the right lower extremity, with pedis artery impalpable on the right side and normal pedis artery on the left side.\nThe color Doppler showed that there was insufficient blood flow with low vascular tension in the right lower extremity, which suggested stenosis or incomplete obstruction proximal to the popliteal artery. Computed tomography (CT) angiography showed that there was complete occlusion of the right common iliac artery and the beginning of internal and external iliac arteries, with no collateral circulation from the contralateral side (), which caused narrowing of arteries of the right lower extremity.\nThe patient underwent an emergency digital subtraction angiography (DSA; ) and catheter-directed thrombolysis combined with balloon angioplasty. The procedure was performed by a contralateral retrograde approach under regional anesthesia. Initially, catheter-directed thrombolysis was performed with urokinase 300,000 units for half an hour. Subsequent angiography showed antegrade flow with large residual thrombi. Then, a balloon was used to get patent recanalization (), followed by urokinase 500,000 units pumped into the right common iliac artery for 24 h. Post-procedure angiography demonstrated smooth blood flow without filling defects in the previously occluded arteries (). The feeling of tingling in the right lower extremity disappeared soon after balloon angioplasty. The right pedis artery got palpable and skin temperature gradually returned to normal. Sensation gradually recovered with disappearing of numbness in the right lower extremity. No presentation of ischemia-reperfusion injury, such as swelling of the right lower extremity, increased creatinine, or hyperkalemia, was detected.\nAfter the procedure, the patient received subcutaneous low-molecular-weight heparin calcium 4100 units twice daily for 2–3 days, warfarin 2.5 mg once daily for half a year, and aspirin 100 mg once daily for life time. The international normalized ratio (INR) was maintained within 2–3. The procedure went uneventfully without any complications, and the patient was fine at the 2-year follow-up. Informed consent was obtained from the patient for publication of this case report. The institutional review board of The Third Hospital of Changsha does not require ethics approval for reporting individual cases. |
A 26-year-old male was injured during a fire show performance. After suffering severe burns, he fell and hit his head. Symptoms of injury manifested 6 months later when the patient performed a sudden head movement and began to display severe tetraparesis. Magnetic resonance imaging (MRI) at this time revealed dislocation of the anterior transligamentous atlas dislocation [].\nThe patient's burns were treated before admission for surgical treatment. Fibrous scarring over the burn areas resulted in rigid fixation of the head and cervical vertebral column into an abnormal position [].\nFunctional radiography of the cervical spine indicated the feasibility of a partially closed reduction of the atlas dislocation. Considering the location of the invaginated odontoid process above the hard palatine line [], a transnasal endoscopic odontoid process resection was selected for surgical treatment.\nThe patient underwent a two-stage surgical treatment. During the first stage of treatment, posterior decompression and fixation were performed under halo immobilization. In the second stage of treatment, an endoscopic approach to the odontoid process was performed. The anterior half ring of the C1 vertebra was exposed and the upper part of the half ring was resected. The odontoid process was located at 2 cm from the posterior edge of the C1 arc. An approach to the odontoid process of the C2 vertebra was performed under radiographic guidance. The odontoid process and upper part of the C2 vertebral body were identified and cleaned. The odontoid process and apical ligament were resected using a surgical drill and Kerrison cutters [Figure and ]. Low-intensity cerebrospinal fluid flow from a dural tear was observed after resection of the odontoid process apex, and apparent pulsation of dura was observed after the decompression was completed []. Dural repair was performed using a hemostatic sponge and biological glue. The glue was also used for fixation of the mucous membrane flap []. Postoperative MRI of the craniovertebral joint is shown in . There were no postoperative complications, and the patient was discharged in good condition 9 days after the surgery. |
A 45-year-old woman complaining of right ear pain was first seen in a nearby hospital. Magnetic resonance imaging (MRI) revealed a tumor occupying the PPS with a size of 43 × 38 × 35 mm, which had the characteristics of a trigeminal nerve sheath tumor (), and the nearby hospital referred the patient to our hospital. Fine-needle aspiration cytology of the tumor revealed spindle or pleomorphic sarcoma. The patient underwent resection of the PPS tumor along with part of the middle constrictor muscle, internal pterygoid muscle, and palatine tonsil because of tumor invasion (), using a median mandibular osteotomy approach. The resected tumor had a size of 80 × 65 × 45 mm. A free rectus abdominis musculocutaneous flap, with a size of 35 × 50 mm after trimming, was transferred to cover the internal carotid artery and to compensate for the oropharyngeal defect (). The lingual artery and the external jugular vein were used as the recipient vessels, and one artery end-to-end anastomosis and one venous end-to-end anastomosis were performed. The transfer of a sufficient tissue volume and the use of the Gehanno method were important for the achievement of optimum velopharyngeal and oropharyngeal function. Total surgical time was 653 minutes and total blood loss was 410 ml. The histopathological examination revealed that the tumor was a carcinosarcoma that arose from the minor salivary gland. A postoperative lower mental gingival fistula occurred after surgery, but with conservative treatment, this complication healed in 3 weeks. Therefore, oral ingestion was started 30 days after surgery. About 9 months after surgery, solitary neck lymph node metastasis and lung metastatic recurrence appeared, even though the patient had received postoperative radiotherapy (60 Gy/30 Fr/51 days), which was the only adjuvant therapy that was used. The neck and lung metastatic recurrences required additional surgery. At present, 21 months after the first surgery, the patient is being followed up on an outpatient basis and has shown no further evidence of disease. |
The first patient is a Caucasian 14-year-old girl from Jaén, in the south of Spain, who presented with severe FVa deficiency (FV:C <1%). The deficiency was diagnosed a few days after she was born, following administration of antibiotics to address a urinary infection which triggered mild gastric hemorrhages. Although her hemogram was normal with a normal platelet count, clotting tests showed abnormal values for both coagulation pathways. A determination of coagulation factor levels revealed a severe deficiency of FVa (<1%). The other clotting factors exhibited normal levels. Screening of the patient’s parents showed that both were FVa deficient (mild phenotype). The patient did not experience significant hemorrhagic episodes until she was 8 months old, when she developed a hematoma in her right upper gingiva. As this occurred precisely when her teeth were erupting, she required administration of fresh frozen plasma (FFP), which resolved the hematoma within 24 h. At the age of one, two further bleeding episodes required a FFP transfusion, which immediately resolved the hematoma. Subsequently the patient was diagnosed with an atrial septal defect and was referred to the pediatric cardiology department. Corrective surgery was indicated at the age of two. Prophylactic FFP was administered preoperatively and one week after surgery. No intraoperative complications occurred, and correction of the cardiac defect was achieved. In the next few years, the patient presented only with mild episodes of epistaxis, which coexisted with upper airway infections. Local and oral administration of tranexamic acid resolved the hemorrhage in all but one of the cases, in which FFP had to be administered to address the epistaxis. At the age of six she presented with left hip pain of 12 hours’ evolution. A diagnostic ultrasonogram revealed hemarthrosis in the left hip and no other previous trauma. Administration of FFP every 12 h during the first 24 h and every 24 h thereafter over 5 days resulted in progressive improvement. At age seven, she was admitted with vomiting, abdominal pain and mild yet progressive anemization. While in hospital, the patient experienced headache. Her family explained that on her birthday she had sustained a fall leading to mild cranial trauma without loss of consciousness. A cranial CT-scan revealed a predominantly dense extra-axial collection in the left frontal region extending to the cerebellar tentorium arising from a left subdural hematoma with a biconvex anterior portion, which suggested the possibility of an associated epidural component. Following examination by the neurosurgical department, it was decided to correct her coagulation disorder and adopt a watch-and-wait approach. Administration of FFP every 12 h for 5 days resulted in a slight resorption of the hematoma. The patient is now 14 years old and in the past 2 years has presented with various bleeding episodes, some of which required admission to hospital and administration of FFP and Octaplas® (14 mL/kg), which was initiated around 3 years ago. |
A 20-year-old woman presented to the emergency department with complaints of severe abdominal pain, multiple episodes of vomiting and obstipation for one day. She was a known case of abdominal tuberculosis and had received anti-tubercular treatment for two months. Vital signs showed a blood pressure of 100/64 mm Hg, heart rate of 110 per minute, respiratory rate of 18 per minute, oxygen saturation of 96% on room air and a temperature of 100.5°F. She was cachectic with a weight of 28 kg which was less than 5th percentile for gender and age matched normal population. Physical examination revealed diffuse abdominal tenderness and rigidity. Her erect abdominal radiography showed gas under right dome of the diaphragm. Clinical diagnosis of perforation peritonitis was made and the patient was wheeled to the operating room for emergent laparotomy. Intraoperatively, a 3 cm x 2 cm single ileal perforation was found 1 foot proximal to the ileocecal junction, with unhealthy bowel margins and multiple mesenteric lymphadenopathy. Resection of perforated bowel segment with proximal ileostomy and mucus fistula of distal ileum was performed. Later, histopathology report confirmed tubercular etiology showing caseous necrosis in mesenteric lymph nodes and epithelioid granuloma in ulcer edge biopsy. Two days after the surgery, the patient was started on oral feeds and was accepting meals orally subsequently. Ten days following surgery, the patient had multiple episodes of bilious vomiting with stoma output decreasing to less than 100 mL per day. Her abdomen remained flat with no increase in bowel sounds. Abdominal radiography showed dilated stomach with no air fluid levels. Oral feeds were stopped and a nasogastric tube was placed for drainage of gastric contents. A contrast enhanced computed tomography (CECT) abdomen was done to determine the cause of obstruction which revealed a decreased aortomesenteric angle of 15° (Figure ).\nAs a result of this decreased angle, an abrupt collapse of the third part of the duodenum was seen along with proximal duodenal and stomach dilation (Figure ).\nBased on the clinical features and CECT findings, diagnosis of SMA syndrome was made and the patient was managed conservatively. The patient was not tolerating enteral feeds and no relief of postprandial abdominal pain was noticed even on changing to left lateral decubitus or prone position. Considering the patient’s nutritional condition, total parenteral nutrition (TPN) was started. Anti-tubercular drugs were continued with oral sips. She was started on 1500 kcal/day (50 kcal/kg/day), taking care of refeeding syndrome and was gradually titrated up to provide maximal caloric support. Monitoring of weight, blood glucose, electrolytes, liver function test, and lipid profile was done to prevent any TPN complications. A good clinical response was seen after starting TPN. The patient’s weight gradually increased to 34 kg in three weeks and gradually the stoma output increased and nasogastric drainage decreased. TPN was tapered off gradually with concurrent starting of oral feeds. On discharge, the patient was advised a high caloric and protein diet with small frequent meals. A follow-up CECT was done after six weeks from discharge which showed an increase in aortomesenteric angle from 15° to 21° (Figure ) with the patient being asymptomatic and a total weight gain of 12 kg post-surgery. |
A 45 years old man presented with one month history of generalized abdominal pain and increasing abdominal distension. Three days prior to admission the pain was worsening in severity with associated nausea and constipation. The patient denied any fever, chills or gastrointestinal bleeding. His prior medical history included chronic pancreatitis, liver cirrhosis, polysubstance use including alcohol and multiple episodes of seizures. The patient had many prior admissions in our hospital for chronic pancreatitis and was diagnosed with a pancreatic pseudocyst and bacterial peritonitis in one prior admission. The patient continued to consume alcohol until the current admission despite alcohol cessation counseling multiple times on prior admissions. Physical examination was remarkable for marked abdominal distension with mild generalized tenderness and shifting dullness. Laboratory findings revealed elevated white blood cell count, abnormal liver function tests and elevated amylase and lipase levels. Contrast CT scan of abdomen revealed acute on chronic pancreatitis with extra-hepatic biliary duct and main pancreatic ductal dilatation. The previously noted pseudocyst was resolved with interval development of large amount of ascites with partial small bowel obstruction. A diagnostic and therapeutic paracentesis was performed. Ascitic fluid analysis revealed a white blood cell count of 2470 cells/mm3 with 98% granulocytes, total protein was > 3g/dL. Ascitic fluid amylase was > 20,000 IU/L which increased to 35,000 IU/L on a subsequent paracentesis. Ascitic fluid culture grew Streptococcus viridans which was treated with antibiotics for two weeks. Patient was managed conservatively including holding oral feedings and octreotide infusion but continued to have worsening ascites and required repeated therapeutic paracentesis. A diagnosis of pancreatic ascites with possible pancreatic duct dehiscence was made. An endoscopic retrograde cholangiopancreatography (ERCP) was performed, which confirmed a duct dehiscence with extravasation of the injected contrast () and was treated with placement of a stent. Patient improved clinically and symptomatically with a repeat paracentesis showing decreased amylase levels. On a follow-up clinic visit, patient was noted to have marked improvement in abdominal distension and discomfort. |
A 30-year-old male (height: 175 cm, body weight: 105 kg) presented with fever for 6 days, and abdominal pain and diarrhea for 2 days. He was admitted to the gastroenterology department of an outside hospital. He did not have any history of underlying disease and had recently stopped smoking. He worked for 2 weeks at another hospital where a patient diagnosed with MERS. His initial vital signs and results of initial laboratory examination were unavailable to us, because he was admitted to an outside hospital. However, according to information from that hospital, physical examination on admission to that hospital revealed normal vital signs (except for fever) and initial laboratory examination showed mild elevation of liver enzymes. In that hospital, he firstly underwent chest and abdominal radiographs and abdominal CT for further evaluation of abdominal pain. The initial chest radiograph, which was taken 6 days after the onset of fever, showed a patchy increased opacity in the left lower lung zone, retrocardiac area (). Abdominal CT showed no specific finding in abdominal and pelvic organs; however, a patchy area of consolidation with an air-bronchogram was detected in the left lower lobe of the lung, which was mainly peripherally located (). Conservative treatment with intravenous fluid was used for abdominal pain. Although he did not initially have any respiratory symptoms, he began coughing 9 days after the onset of fever. Because of this new respiratory symptom, occupational history, and a recent MERS outbreak in Korea, he underwent laboratory examination for MERS. A follow-up chest radiograph, which was taken 10 days after the onset of fever, showed newly developed patchy areas of ill-defined increased opacity in the right upper lung zone and increased extent of the previously detected consolidation in the left lower lung zone (). A chest CT scan performed on the same day showed multifocal patchy areas of nodular consolidations orgwith ground-glass opacity (GGO) halo and nodular GGO lesions in both upper lobes, which were mainly slightly peripherally located (). Moreover, this scan also demonstrated increased extent of mixed consolidations and GGOs with air-bronchograms in both lower lobes, mainly in the dependent area (). There was no pleural effusion or significant lymph node enlargement. Finally, he was diagnosed with MERS by testing the sputum by real-time reverse-transcriptase-polymerase chain reaction (rRT-PCR) (). For intensive care and management of MERS, he was transferred to a negative pressure room of our hospital on the day of diagnosis. Physical examination in our hospital showed fever (38.4℃) and elevated blood pressure (154/106 mm Hg). His vital signs on admission are summarized in . Laboratory examination on admission of our hospital showed normal white blood cell counts (5640/µL), and elevated serum aspartate aminotransferase (AST, 155 international unit [IU]/L), alanine aminotransferase (ALT, 77 IU/L), lactate dehydrogenase (904 IU/L), and C-reactive protein (CRP, 9.39 mg/dL).\nHe was administered antiviral agents (ribavirin, kaletra [ritonavir + lopinavir], and interferon-alpha) and antibiotics (tabaxin [piperacillin + tazobactam] and cravit [levofloxacin]). Daily follow-up radiographs were taken to evaluate disease progression. Fever disappeared after 2 days of treatment; although radiologic abnormalities remained on early follow-up radiographs, then their extent decreased after 5 days of treatment. After a week of treatment, he showed no symptom. Follow-up laboratory examination showed normalization of CRP (0.14 mg/dL), slightly deceased but remained elevated AST (131 IU/L), whereas increased ALT (142 IU/L). Finally, follow-up sputum tests by rRT-PCR (conducted twice) showed negative results, and he was discharged after 13 days in our hospital.\nOn the day of discharge (23 days after the onset of fever) he underwent chest radiograph and chest CT scan. The chest radiograph depicted markedly decreased extent of previous increased opacities in the right upper and left lower lung zones (). The chest CT scan showed markedly decreased extent of previously detected lesions with residual nodules or GGOs in both lungs, and developed traction bronchiectasis within retracted consolidation with volume loss in the left lower lobe, which suggested fibrosis (). |
A 55-year-old male was referred to our department for total abdominal aorta obstruction with intermittent claudication. At first, he visited the emergency room with epigastric pain. He had a history of hemigastrectomy and gastrojejunostomy for acute gastric ulcer with perforation 4 years ago. He also had a history of diabetes mellitus and 30 pack-years smoking. Abdominal sonography showed the pancreas of diffuse enlarged parenchyme with normal common bile duct. Abdominal computed tomography (CT) showed the swelling of pancreas and small amount of fluid retention around pancreas with no demonstrable bile duct stone and total occlusion of infrarenal aorta to both common iliac arteries. After he completed antibiotics therapy for acute pancreatitis and cholangitis, he was transferred to our department for treatment of total occlusion of abdominal aorta ().\nCT angiography showed total occlusion of infrarenal aorta, both common iliac arteries and left external iliac artery with collateral arterial flow into both femoral arteries. CT angiography also showed focal short segmental stenosis in superior mesenteric artery origin area with abundant collateral flow. Preoperative ankle-brachial indices were 0.49 and 0.42 (right and left). Considering relatively young age of the patient, durable bypass procedure like aortobifemoral artery was needed. However, the patient has previous abdominal surgery for gastric ulcer perforation and recent inflammatory conditions, pancreatitis and cholangitis. After discussing various surgical alternatives, we decided to perform descending thoracic aorta to both femoral artery bypass surgery to avoid operation in a hostile abdomen.\nWith double-lumen endo-tracheal intubation to allow deflation of the left lung, the patient was positioned with the right semi-lateral position. The pelvis was placed flat to allow access to both groins. Left posterolateral thoracotomy was performed through the eighth intercostal space, then, the distal descending thoracic aorta just above diaphragm and both femoral arteries were exposed simultaneously. The distal descending thoracic aorta was mobilized from the parietal pleura to allow placement of a partially occluding clamp and a stab wound was made for graft. Leaving the inguinal ligament intact, the oblique and transverse muscles of left side were split and the left retroperitoneal space was entered. Using blunt finger dissection, retroperitoneal tunneling was performed between the left hemithorax and the left suprainguinal preperitoneal space and cross over tunnel between both groin incisions was created similarly. After partial clamping on thoracic descending aorta with systemic heparinization, a 10 mm polytetrafluoroethylene (PTFE) graft (Gore-Tex; Flagstaff, AZ, USA) with externally supported rings was anastomosed to the aorta proximally in an end-to-side fashion. And then distal anastomosis (left groin) was performed to left common femoral artery. After that, right common femoral artery and 10 mm graft were connected with 8 mm PTFE graft with rings. A chest tube (28Fr) was placed in the left pleural space and all incisions were closed.\nPostoperative course was uneventful and postoperative ankle-brachial indices were improved from 0.49 and 0.42 to 0.95 and 0.94 (right and left). All bypass grafts were patent in postoperative CT angiography (). The patient was discharged without any complication and claudication 7 days after surgery. He is doing well without symptom at 30 months postoperatively. |
A 20-year-old woman presented to the emergency department with complaints of severe abdominal pain, multiple episodes of vomiting and obstipation for one day. She was a known case of abdominal tuberculosis and had received anti-tubercular treatment for two months. Vital signs showed a blood pressure of 100/64 mm Hg, heart rate of 110 per minute, respiratory rate of 18 per minute, oxygen saturation of 96% on room air and a temperature of 100.5°F. She was cachectic with a weight of 28 kg which was less than 5th percentile for gender and age matched normal population. Physical examination revealed diffuse abdominal tenderness and rigidity. Her erect abdominal radiography showed gas under right dome of the diaphragm. Clinical diagnosis of perforation peritonitis was made and the patient was wheeled to the operating room for emergent laparotomy. Intraoperatively, a 3 cm x 2 cm single ileal perforation was found 1 foot proximal to the ileocecal junction, with unhealthy bowel margins and multiple mesenteric lymphadenopathy. Resection of perforated bowel segment with proximal ileostomy and mucus fistula of distal ileum was performed. Later, histopathology report confirmed tubercular etiology showing caseous necrosis in mesenteric lymph nodes and epithelioid granuloma in ulcer edge biopsy. Two days after the surgery, the patient was started on oral feeds and was accepting meals orally subsequently. Ten days following surgery, the patient had multiple episodes of bilious vomiting with stoma output decreasing to less than 100 mL per day. Her abdomen remained flat with no increase in bowel sounds. Abdominal radiography showed dilated stomach with no air fluid levels. Oral feeds were stopped and a nasogastric tube was placed for drainage of gastric contents. A contrast enhanced computed tomography (CECT) abdomen was done to determine the cause of obstruction which revealed a decreased aortomesenteric angle of 15° (Figure ).\nAs a result of this decreased angle, an abrupt collapse of the third part of the duodenum was seen along with proximal duodenal and stomach dilation (Figure ).\nBased on the clinical features and CECT findings, diagnosis of SMA syndrome was made and the patient was managed conservatively. The patient was not tolerating enteral feeds and no relief of postprandial abdominal pain was noticed even on changing to left lateral decubitus or prone position. Considering the patient’s nutritional condition, total parenteral nutrition (TPN) was started. Anti-tubercular drugs were continued with oral sips. She was started on 1500 kcal/day (50 kcal/kg/day), taking care of refeeding syndrome and was gradually titrated up to provide maximal caloric support. Monitoring of weight, blood glucose, electrolytes, liver function test, and lipid profile was done to prevent any TPN complications. A good clinical response was seen after starting TPN. The patient’s weight gradually increased to 34 kg in three weeks and gradually the stoma output increased and nasogastric drainage decreased. TPN was tapered off gradually with concurrent starting of oral feeds. On discharge, the patient was advised a high caloric and protein diet with small frequent meals. A follow-up CECT was done after six weeks from discharge which showed an increase in aortomesenteric angle from 15° to 21° (Figure ) with the patient being asymptomatic and a total weight gain of 12 kg post-surgery. |
A 40-year-old female presented with chronic headache with infrequent exacerbations. She presents with worsening headache for three months with associated vertigo, nausea, and vomiting not responding to analgesics or vestibular sedatives. Her symptoms initially started twelve years ago as a diffused mild headache, which persisted through the day. Gradually, the headache worsened to a severe headache episodically associated with vertigo, nausea, and vomiting. These episodes lasted for two to three days and got resolved. She was treated with flunarizine for suspected basilar migraine but did not show any response. From the last year, she had monthly exacerbations of headache associated with distressing vertigo, unsteadiness of gait, and right-sided body numbness. In between these episodes, she had a significant dull diffuse headache not responding to simple analgesia. She did not complain of fever or night sweats but had constitutional symptoms lasting for several months. All of these symptoms severely affected her daily activities and functionality.\nDuring the last 17 years, she had repeated episodes of neurological deficits. Even before the headache appeared, she has presented with visual impairment of the right eye and right lateral rectus palsy and was treated as retrobulbar neuritis with good response to methylprednisolone. One year later, she developed left-sided visual impairment, which fully responded to methylprednisolone. MRI imaging at that time revealed normal results. Few months after this event, she got admitted with right hemisensory loss with hemiplegia, and a demyelination disease or hemiplegic migraine was suspected. Second MRI was performed at this admission, and no abnormalities were detected again. Eight years ago, she had developed a left lower motor type facial nerve palsy, which was attributed to Bell's palsy. Within the last year, she was diagnosed to have depression and anxiety for which she was treated for few months. Other than the first two instances, she was not treated with steroids thereafter. She did not complain of weight loss and did not have constitutional symptoms or chest symptoms during these periods.\nOn examination, she is an averagely built female with a BMI of 23 kg/m2. She is afebrile, pale, and did not have lymphadenopathy. Her GCS was 15/15, and she was conscious and rational with normal pupillary response, visual acuity, visual field examination, and fundoscopy. There was no neck rigidity, and she had residual left lower motor VII palsy. She had an ataxic broad-based gait with unsteadiness. Upper and lower limb examination is clinically normal. Her respiratory, cardiovascular, and abdomen examinations were unremarkable.\nInvestigations revealed a hemoglobin count of 9 g/dL with normal white cells and platelets. ESR was elevated to 86 mm/1st hour. Renal- and liver-related biochemical investigations were normal with an alkaline phosphatase within the normal range. Initial MRI scans of the brain done 10 years ago did not reveal any abnormalities such as demyelination, optic nerve enhancement of focal lesions in the cerebrum, or cerebellum. CSF examination revealed an elevated protein level of 55 mg/dl with normal glucose and cells with negative oligoclonal bands or TB-PCR. Vasculitis investigations including ANA, ANCA (ELISA and Immunofluorescence), and RF were negative. Syphilis serology and HIV testing were also negative. Chest radiograph, ultrasound abdomen, and CT scan of chest and abdomen did not reveal any mediastinal lymphadenopathy or focal lesions in visceral organ or evidence of any malignancy. Serum ACE levels (19 µ/l) and ionized calcium levels were normal. We performed a new MRI scan of the brain with contrast, which revealed a diffuse and patchy meningeal thickening and enhancement mainly in the right frontoparietal and left occipital regions with a minor enhancement of bilateral optic sheaths (). Her NMO antibodies were normal, and the MRI did not show any areas of demyelination. Therefore, she underwent a dural biopsy from the thickened dura, which revealed large areas of caseous necrosis surrounded by epithelioid histiocytes and lymphoid cells with a few isolated giant cells in the adjacent vicinity (). There were no features of vasculitis or sarcoidosis. TB-PCR of tissue and acid-fast bacilli were negative. Final conclusion was necrotizing granulomatous inflammation suggestive of dural tuberculosis. This diagnosis was presumed by the presence of necrotizing granulomatous necrosis with caseation with a strongly positive Mantoux test of 25 mm (), and later was supported by a marked response to antituberculous medication.\nWe initiated her on antituberculous therapy (all four drugs for 3 months and 9 months of rifampicin and isoniazid) without streptomycin as she is already having vestibular symptoms. Steroids were added concurrently (1 mg/kg) and was continued for 6 weeks and was tailed off over a month. She experienced a marked improvement of her headache, and she could do her daily activities normally. After a year of anti-TB medication and follow-up, she did not complain of any worsening of symptoms. |
A 55 year old man was diagnosed with anemia by his family physician towards the end of 2017. In February of 2018, an upper GI endoscopy and colonoscopy were performed which showed a right colon cancer. CEA was 40.4. CT of the chest, abdomen, and pelvis was obtained in February of 2018. This showed a mass 7 cm in greatest diameter that was immediately adjacent to the undersurface of the right liver and contiguous with numerous loops small bowel and the lowest part of the second portion of the duodenum (). There were mildly prominent mesenteric lymph nodes but no evidence of distant metastatic disease.\nIn February of 2018, the patient underwent a single port laparoscopic right colon resection. The surgeon described the tumor as stuck to the undersurface of the liver with adhesions taken down without difficulty. On histopathologic examination, the tumor was PT3N0M0 with 0/16 positive nodes. Perineural invasion was identified. The cancer was moderately to poorly differentiated. Systemic chemotherapy was not recommended.\nIn June of 2018, the patient began noting problems with digestion and pain after eating. CEA had increased to 940 ng/ml. Repeat CT scan showed multiple nodules within the greater omentum compatible with peritoneal metastases. The liver, kidneys, and ureters were normal. There was marked stranding within the right colon resection site (). Bowel loops proximal to the prior ileocolic anastomotic site were moderately dilated and fluid-filled. A mass was noted in the abdominal wall at the laparoscopic port site. There was also a mass in the rectovesical space immediately adjacent or invading the right and left seminal vesicles.\nIn July of 2018, the patient underwent exploratory laparotomy. The posterior surface of the liver was layered by cancer. The undersurface of the right hemidiaphragm was covered by tumor nodules. The pelvis contained a 4 cm tumor mass invading into the seminal vesicles. The old ileocolic anastomosis was involved and needed to be resected. The umbilical port site was infiltrated by cancer. Near complete resection was possible by cytoreductive surgery.\nIntraoperatively, the patient was treated with HIPEC-melphalan []. This postoperative course was unremarkable. The histopathology showed adenocarcinoma compatible with the primary tumor in all tissues submitted. The peritoneal cancer index was 28 and the CC score was 2 []. The resection was judged to be a palliative effort and its major goal was to relieve intestinal obstruction and to prepare the patient for rapid initiation of palliative systemic chemotherapy.\nNeither of our patients was evaluated by the MDT preoperatively. Their preoperative and intraoperative management was evaluated in an attempt to improve the outcome of subsequent patients. |
A 74-year-old female was referred to our department of surgery with malodorous fecal vaginal discharge that had lasted several weeks. Medical history included multiple episodes of acute diverticulitis and dementia. She had no abdominal pain or fever. Upon admission, her laboratory tests were within the normal range. Pelvic examination revealed an atrophied vagina and malodorous yellowish vaginal discharge. A colovaginal fistula, secondary to diverticular disease, was suspected as it is the most common type of fistula between the gastrointestinal and female reproductive system. Cultures of the vaginal discharge were sent. Nil per os, total parenteral nutrition, and intravenous antibiotic therapy with ciprofloxacin and metronidazole were applied to the patient until a definitive diagnosis would be made. An abdominal computed tomography (CT) showed multiple diverticula and findings indicative of chronic diverticulitis, such as thickening of the wall of the sigmoid colon and pericolic fat stranding (Figures and ). A colonoscopy was performed, revealing diverticula of the sigmoid colon. Magnetic resonance imaging (MRI) revealed a fistula between the sigmoid colon and the uterine fundus and air bubbles in the uterine cavity and the vagina (). Surgical management was decided. Intraoperative findings included a colouterine fistula side to side to a jejunouterine fistula as well (). Inflammatory adhesions of the sigmoid colon and the jejunum to the wall of the uterus, as a result of the repeated episodes of diverticulitis, were found, explaining the presence of the fistulae, as well as adhesive lesions between the urinary bladder and uterus. Taking in mind the age of the patient and the intraoperative findings, we performed a total abdominal hysterectomy with bilateral oophorectomy and en bloc resection of the defective part of the sigmoid colon and jejunum (Figures and ). Primary jejunojejunal and colocolonic anastomoses were performed. During surgery, a bladder injury was recognized and repaired immediately. Postoperatively, the patient had a fever because of an urinary tract infection which was treated with antibiotics. Rest of the postoperative course was uneventful, and the patient was discharged on the 21st postoperative day. |
The patient was a 65-year-old woman without notable antecedents presented to our institution for progressive left hip pain for approximately 8 months. It was a mechanical pain of the hip well relieved by the usual analgesics. The appearance of walking distance and the poor response to analgesics forced her to consult in our center.\nThe BMI was 35,5. The walk was almost normal. There was no cutaneous scar on the lateral side of the left hip or on the ipsilateral buttock. There was a good trophicity of the abductors. Lateral rotation and abduction were markedly diminished. The rest of the exam was strictly normal. The pelvis AP () and lateral () left hip radiographs revealed signs of hip osteoarthritis. We concluded that it was a symptomatic left hip osteoarthritis that was more and more disabling in an obese woman of 71 years with no particular history. We indicated THA by posterolateral approach.\nIn the operating room, after the skin incision and subcutaneous haemostasis, we discovered in the adipose tissue about 5 cm thick a kind of well-circumscribed shell of about 2.5 cm of axis. Her incision gave rise to a whitish, thick color, looks a little oily collection (), resembling a purulent collection (). A sample for bacteriological investigation in a lab was carried out. The hull with its clear boundaries within the gluteal fat was resected and entrusted to the pathologist. All the neighborhood tissues were healthy (very localized lesion).\nIn front of this collection which appeared to be purulent, we limited ourselves to the resection of this hull, the cleaning of the wound, and the deferred implantation of the prosthesis.\nCytobacteriological examination of the specimen revealed its greasy appearance, epithelial and lymphocytic cells; there were no visible germs. Histological examination of the resected shell revealed a fibrous wall with chronic inflammatory remodeling made of lymphocytes and plasma cells with no necrosis centers.\nIn the light of these laboratory results, we conducted the interview of the patient, who reported a notion of malaria for about two months to the screen treated with an intramuscular injection on the right buttock of the compounds derived from artemisinin. We found the result of the thick drop before the injection which was positive and that of the injection which had not been negated; the patient was then successfully treated orally. The sample was sent to a lab for confirmation by artemether identification by thin layer chromatography (TLC).\nA sample of 40 g of human fat was treated with ethyl acetate (50 ml × 3) after filtration on Whatman paper, the solvent was evaporated, and the residue was taken up with acetone (40 ml) constituting the sample to be analyzed. Artemether was purchased from a local pharmaceutical company.\nImplementation of the TLC: solution to be analyzed: 20 μl of sample; control: artemether (80 mg/mL), 10 μl deposit; support: silica gel GF254; mobile phase (10 ml): dichloromethane, ethyl acetate (7/3); and developer: 25 ml anisaldehyde reagent, 5 ml concentrated acetic acid, 450 ml ethanol, and 25 ml concentrated sulfuric acid. Using a capillary tube, 20 μl of the sample was deposited on the plate (silica gel GF254), the control 10 μl. The plate is placed in a tank previously saturated with the migration or elution solvent (mobile phase) which covers the bottom of the tank at 5 mm height. The migration of eluting solvent causes the substances contained in the samples at various speeds; spots are formed characterizing the substances present in the sample.\nThe plate was removed from the tank as soon as the solvent front reached about 9 cm. The plate was dried and observed under a UV lamp at 254 nm and then revealed with the developer which will characterize the artemether in human fat.\nThe plate then shows an orange spot on the left side of the sample and a spot with the same color on the right side of the control; the two spots have the same front report as shown in . This indicates that there was artemether in this human fat sample. |
A 38-year-old male with a medical history of HBV-associated PAN presented with acute abdomen and septic shock. He was diagnosed with PAN 6 months earlier and was under treatment with prednisolone (32 mg daily) and cyclophosphamide (150 mg every other day). The patient was also prescribed the antiviral agent Tenofovir, but stopped taking it 2 months prior to the incident, against medical advice. The patient's medical history also included chronic renal failure and diabetes mellitus Type II. On work up, a standing chest radiograph revealed free sub diaphragmatic air.\nDuring laparotomy, peritonitis was found due to three perforations of the small intestine. Segmental enterectomy with anastomosis was performed at two sites, the first at 10 cm from the ligament of Treitz, and the second at about 1 m from the ileocecal valve.\nThe patient was then transferred to the Intensive Care Unit on mechanical ventilation and circulatory support, in acute-on-chronic renal failure. On the third postoperative day he was weaned off the ventilator and remained haemodynamically stable. After consultation with the rheumatologist, the patient started treatment with tenofovir orally and IV methylprednisolone 1 gr daily for 3 days.\nOn the seventh postoperative day the patient's abdominal drain catheter presented enteric content, so he underwent a second explorative laparotomy. Two new perforations and multiple areas of patchy necrosis along the whole length of the small intestine were found (Fig. ). All the lesions were suture repaired after debridement, and the patient was left with an open abdomen using a vacuum device, with a plan for re-laparotomy in 48 h (Fig. ). After the second operation the patient started plasma exchanges for the treatment of PAN, along with IV cyclophosphamide 500 mg stat and IV methylprednisolone 1 gr daily for 3 days and then IV prednisone 60 mg daily.\nOn the third laparotomy, three new necrotic lesions, two on the small intestine and one on the cecum, were found and suture repaired (Fig. ). A necrotic lesion on the left lobe of the liver was also found (Fig. ). On the fourth laparotomy, segmental enterectomy with anastomosis and cholecystectomy were performed due to anastomotic leak and a gangrenous gallbladder.\nThe patient died 15 days after the first operation, despite aggressive treatment, due to septic shock and multiple organ failure. |
A seven-year-old Korean girl presented with left knee pain of three months' duration. A physical examination demonstrated a five-degree extension block and tenderness on the lateral joint line. The result of a McMurray test was positive. An MRI scan revealed a complete discoid lateral meniscus with a bucket handle tear. On arthroscopy, a complete discoid lateral meniscus with longitudinal tear was found that extended throughout the entire meniscus. Subtotal meniscectomy with reshaping of remnant meniscus tissue was performed. Our patient had no further symptoms stemming from the torn meniscus and recovered a full range of motion. Activity was not restricted following recovery from the surgical intervention.\nTwo years after her first operation, our patient presented with a snapping sound and intermittent pain involving her right knee. A physical examination at this time revealed mild tenderness to the lateral joint line, but all other test results and findings from plain radiographs were normal. An MRI scan showed a complete discoid lateral meniscus with a 1.5 by 1.5 cm osteochondral lesion involving the posterior articular surface of the lateral femoral condyle (Figure ). There was no evidence of fluid signal intensity between the host and fragment on a T2-weighted MRI scan (Figure ). Initially, our patient was treated with conservative management consisting of activity modification. However, our patient had persistent symptoms despite six months of conservative management and she therefore underwent operation. On arthroscopy, a complete discoid lateral meniscus was identified (Figure ). The articular surface of the lateral femoral condyle had normal articular continuity and contour, but softening of cartilage at the margins of the OCD within the lateral femoral condyle without breach or fibrillation was found. We performed meniscoplasty that provided a stable 6 mm peripheral of the remaining meniscus and no treatment was performed for the OCD lesion (Figure ). Post-operatively, our patient was allowed to begin full weight bearing without immobilization and started a physical therapy protocol to improve the range of motion in her knee. Five months after the operation, an MRI scan demonstrated complete resolution of the previous OCD lesion of the lateral femoral condyle (Figure ). There was no restriction of early activity following the surgical intervention. Our patient had no symptoms on either knee and had returned to full daily activity. |
A one month old baby girl was brought by her mother to the lactation clinic for further evaluation due to the difficulty of breastfeeding. The mother complained of pain in the nipple which was present throughout the entire breastfeeding session and usually recurred with each episode of breastfeeding. Two weeks after delivery, the mother noticed a tooth on the baby’s lower left gum. A week later, she noticed an ulcer under her baby’s tongue, which occasionally bled.\nIn light of the continuous pain during breastfeeding, the mother became reluctant to continue and infant formula milk was given as a substitute. Self-examination by the mother did not reveal any nipple crack or breast injury. Her antenatal history was uneventful. There was strong family history of natal teeth, and this baby’s siblings had similar problems.\nOn examination, the mother’s breast appeared to be normal and consistent with a lactating breast. There was no inflammation or engorgement. Some mild eczema was noted around the nipple, but there was no infection. Cervical and axillary lymph nodes were not palpable.\nExamination of the oral cavity of the baby revealed a neonatal tooth over the left anterior region of the mandibular ridge. The tooth measured 2 mm × 1 mm in size, was whitish opaque, and had Grade II mobility (Fig. ). There was also a whitish ulcer over the ventral aspect of the tongue measuring 1 mm × 1 mm (Fig. ). The parents declined radiological investigations and tongue biopsy for the baby. Hence, the clinical diagnosis was neonatal tooth associated with Riga-Fede disease causing difficulty in breastfeeding.\nThe baby was then referred to the pediatric dental surgery department. After a thorough discussion with the parents, the neonatal tooth was extracted under local anesthesia. The tooth had a crown but lacked a root. Following extraction, the baby did not have any complication such as bleeding and infection. The wound healed well within 2 days and she successfully resumed taking breast feeds.\nCurrently, at 10 months post-extraction, the child is growing well and still breastfeeding. Examination of the oral cavity revealed that only one central lower incisor tooth was present (Fig. ). |
A 38-year-old woman with schizophrenia committed suicide by a self-infliction of sharp force into the chest and abdomen. She presented to our hospital in a critical condition. On arrival in the emergency room (ER), she developed hemorrhagic shock, which leads to an emergent abdominal damage control surgery under general anesthesia. She lost cardiac activity in the operating room during exploratory laparotomy. A left-sided basal thoracostomy, cross-clamping of the descending thoracic aorta, and open cardiac massage were also performed. She received gauze packing at the bleeding point and temporarily close the abdomen and chest wall. After the surgery, she was transferred to the intensive care unit with mechanical ventilation and recovered from hemorrhagic shock. Two days later, a second-look laparotomy was performed. ER doctors verified the hemostasis and closed the patient’s abdomen and chest. On the same day, a chest radiograph revealed pulmonary atelectasis of the left upper lobe (). Computed tomography (CT) revealed alveolar bleeding, pneumonia and atelectasis of the left upper lobe (). Despite antibiotic therapy and repeated endobronchial sputum aspiration, radiologic findings were not improved. On the seventh postoperative day, she was referred to our department for suspected lung abscess. When compared to the chest CT images on the day of initial surgery, pulmonary torsion was suggested (). The sagittal CT images were reconstructed, and the torsion of the left upper lung lobe was diagnosed (). Therefore, an emergency left upper lobectomy was performed through a left thoracotomy. Intraoperatively, the left upper lung lobe was rotated clockwise and was hemorrhagically and congestive infarcted. Due to severe pulmonary congestion, it was difficult to obtain the surgical field of view. The pericardial sac was opened and the intrapericardial isolation of the left upper pulmonary vein was performed. To prevent separation of the thrombus and necrotic material in the left upper lobe, we first dissected the left upper pulmonary vein before resolving the torsion and performing the lobectomy. She had a complete pulmonary fissure. Moreover, only one first pulmonary artery branch had ruled the approximately pulmonary arterial flow of her left upper lobe. The left upper lobectomy was performed. The patient made an uneventful recovery. |
A 41-year-old female with obesity and polycystic kidney disease is referred to the cath lab after an episode of chest pain with troponin elevation one week after primary PCI in the context of inferior acute myocardial infarction with ST elevation. Angiography at the time revealed a balanced-dominant circulation, with TIMI flow 3 in the right coronary artery (RCA) without stenotic lesions and a subocclusive lesion in a small posterolateral branch (PLB) from the circumflex artery which was considered to be the culprit lesion. Balloon angioplasty was performed with good final result and TIMI 3 flow.\nIn the present episode angiography showed persistent good result of the previous PCI, and total occlusion of the proximal RCA, that was suspected to be caused by spontaneous dissection because of the presence of haziness at the site of the occlusion and disappearance of several acute marginal branches as compared with the previous exam. We then decided to perform OCT (Terumo Lunawave® OFDI system) that clearly revealed the double-lumen morphology and also that the guidewire was in the false lumen (Figures and ). We then proceeded to angioplasty under OCT guidance. With live OCT images, without recording an actual pullback, we used small flushes of contrast until we were able to confirm that the guidewire was on the true lumen (Figures and ). The OCT could also identify entry tear very clearly (). Once the true lumen was secured, we proceeded to angioplasty with a drug-eluting stent sealing the entry tear in the proximal right coronary artery (). There was final TIMI 3 flow, and the acute marginal branches were again visible, but there was a narrowing in the distal vessel where the false lumen was still visible but showing signs of thrombosis (). To avoid making a full metal jacket, we decided to accept this result and perform a control angiography one month later. After one uneventful month, follow-up angiography revealed persistence of a large dissection that extended to the distal vessel. There was an important compromise of the true lumen by the false lumen. Given the previous STEMI presentation and the negative angiographic evolution within a month, we carried out an OCT that confirmed the dissection and showed that the guidewire was now in the true lumen (Figures and ). In this setting, we decided to seal the dissection with implantation of two more drug-eluting stents in the mid and distal RCA to guarantee the long-term patency of the vessel (). There was residual dissection in the very distal segment, which was a small vessel. |
A medically free 14-year-old male who was obese sustained an injury to his left knee after jumping from 3 stairs. The patient mentioned that he predominantly landed on his left lower limb with his left knee in full extension and in external rotation. The patient started complaining of left knee pain limiting his range of motion and ability to bear weight immediately after the fall. He was brought to the Emergency Department (ER) of King Saud Medical City (KSMC) by his parents immediately after the injury. On physical examination, the left knee was profoundly swollen and bruised. There was tenderness over the tibial tuberosity and lateral joint line. He was unable to actively move the knee joint. The passive range of motion was painful. There were no signs indicating compartment syndrome or neurological or vascular injury. X-ray radiographs revealed a Watson-Jones type IIIB avulsion fracture of the tibial tuberosity apophysis (). A CT scan showed a step of the articular surface more than 2 mm extending to the posterior-lateral epiphyseal part of the proximal tibia (lateral tibial plateau) ().\nThe patient was admitted and was prepared for operative management. A procedure was planned and done on a radiolucent table under general anesthesia. A tourniquet was used to avoid excessive bleeding during the procedure. The tourniquet was inflated after pulling down the quadriceps to avoid blocking the reduction due to the extensor mechanism. The leg is prepped and draped according to the standard orthopedic protocol. Anterolateral approach of the knee was used with an incision starting from the lateral upper border of the patella to 10 cm down. Deep fascia was opened anterior to the iliotibial tract. The fracture line was identified; the reduction of the articular step was done using a reduction clamp and assured using a portable image intensifier on flexion and extension of the knee without arthrotomy. We avoided arthrotomy of the joint to not make it vulnerable to infection and possible scarring. Stabilization of the reduction was maintained using a k-wire. Definitive fixation was achieved with three 3.5 mm partially threaded cancellous screws placed under fluoroscopic guidance for the tibial tuberosity fracture. A proximal tibial plate was slid laterally and was used to buttress the lateral tibial column. Careful placement of the screws was done to not cross the physis with the help of a C-arm (). After fixation, good hemostasis was achieved, drain was placed, and the range of motion was assessed which was full. Closure was done layer by layer, then dressing after. The postoperative plan was to immobilize the knee in a cylindrical cast for 3 weeks with no weight bearing on the left lower limb with the use of crutches for ambulation.\nPostoperative knee CT scan is requested to ensure that the fracture is anatomically reduced. The patient received analgesia and antibiotics, and drain was removed 24 hours post-op. The patient was seen in an orthopedic clinic after 3 weeks, there were no signs of surgical site infection, and the clips were removed. The controlled range of motion was advised using a hinged knee brace throughout the day for 4 weeks. A follow-up X-ray () shows that the fracture is aligned with no loss of reduction or displacement. Physiotherapy is advised 7 weeks postoperative management focusing on the range of motion and strengthening. The patient was seen 6 weeks later, he had full range of motion with no deformity, and there were no complaints reported by the patient like locking or pain. |
A 52-year-old woman was referred to the outpatient clinic of rheumatology department with a 6-month history of dyspnea on exertion [New York Heart Association (NYHA) functional class II to III]. About 20 years ago, she visited a local clinic for non-healing toe wound and had a tentative diagnosis of TA. She had a previous history of vascular surgery for left carotid artery. Ten years later, she visited another local clinic with a complaint of dyspnea on exertion (NYHA functional class II). At that time, echocardiographic examination showed severe pulmonary hypertension with severe tricuspid regurgitation, based on which the attending physician prescribed diuretics and digoxin with a diagnosis of idiopathic pulmonary arterial hypertension. Despite the medications, however, she could not feel any improvement with regard to exertional dyspnea and she was advised to undergo surgery for severe tricuspid regurgitation. During preoperative work-ups, complete occlusion of right main pulmonary artery was incidentally detected. For further evaluation, she was referred to our hospital.\nOn physical examination, her blood pressure was 78/39 mmHg at right arm and 99/70 mmHg at left arm. Her chest radiography showed a prominent left hilar opacity and right atrial enlargement (). Her electrocardiogram showed right atrial enlargement and right ventricular hypertrophy. Laboratory parameters included increases in erythrocyte sedimentation rate (77 mm/hr) and C-reactive protein (2.14 mg/dL).\nOn echocardiography, right ventricle (RV) was severely dilated, its wall thickness was increased, and RV systolic function was decreased. Severe tricuspid regurgitation and severe pulmonary hypertension with maximal pressure gradient of 131 mmHg were noted, as well. On basal parasternal short axis view, the blood ejected from the RV to the main pulmonary artery appeared not to cross the right pulmonary artery. When we carefully traced the proximal right pulmonary artery, we could not clearly delineate its opening (). Lung perfusion scan showed the total absence of perfusion in the right lung with partial perfusion defects in the left lung (), but no pulmonary thromboembolism was clearly demonstrated on computed tomographic pulmonary angiography. The occlusion of right main pulmonary artery and left main pulmonary artery stenosis at the entrance were also clearly demonstrated on computed tomographic angiography (). According to the result of cardiac catheterization performed elsewhere, the mean right atrial pressure was severely elevated (18.4 mmHg), and catheter was impossible to go into the right pulmonary artery. Coronary angiography demonstrated a huge left circumflex coronary artery to right pulmonary artery fistula (). Arch aortography revealed total occlusion of the left subclavian and left common carotid artery in their proximities. Right subclavian artery showed near-total occlusion, as well (). The diagnosis of TA was established based on the criteria of American college of Rheumatology for classification of TA; age at diagnosis (less than 40 years), difference in systolic blood pressure between arms (exceed 10 mmHg), arteriogram abnormality.\nThe patient was treated with corticosteroid, methotrexate, bosentan and symptomatic medications for right-sided heart failure. She is now being followed in the rheumatology clinic on a regular basis and her symptoms showed slight improvement with these medications. |
Our patient was a 35-year-old Norwegian man who suddenly collapsed in the street. Emergency services attended and started cardiopulmonary resuscitation on site, eventually achieving return of spontaneous circulation. The patient experienced another cardiac arrest in the emergency room at the hospital and was given thrombolytic treatment, but he died shortly thereafter. A 12-lead electrocardiogram showed irregular rhythm with both left and right bundle branch block (Fig. ). Apart from elevated lactate, his laboratory test results were normal (Table ). According to the patient’s relatives, he had been healthy without known risk factors or a family history of coronary disease. He had occasionally smoked cannabis and hashish and drank alcohol. The use of other narcotic substances was unknown to his relatives or his general practitioner. The patient had been of medium build with a body mass index of 25 kg/m2. He had experienced slight chest discomfort 1 month before his death; that episode lasted a couple of hours. The autopsy revealed a 20-mm-long thrombus lodged in the left anterior descending (LAD) branch of the coronary artery associated with moderate atherosclerosis in the same segment. The rest of his vascular system showed only focal mild atherosclerosis. His heart weighed 350 g and was not enlarged. The posterior wall of the left ventricle and part of the septum showed a sharply demarcated infarcted area with a largest diameter of 8 cm. It was yellow-red, soft, and had reddish edges. The rest of the heart was unremarkable. The left and right ventricle walls were 10 and 5 mm thick, respectively.\nOther autopsy findings were the main diagnoses of acute MI and thrombosis of the LAD branch of the coronary artery as well as the secondary diagnoses of pulmonary atelectasis; blood stasis in the liver, spleen, and kidney; and mild focal atherosclerosis in the aorta and its major branches. The histological specimens from the heart and LAD branch were fixed in 4 % formaldehyde, embedded in paraffin, and cut at 4 μm. Sections were stained with hematoxylin and eosin. Sections from the heart showed infarction with early organization (7–10 days old) and acute reinfarction at the edges (Fig. ). At 7–10 days old, an infarction is maximally yellow-tan and soft, with depressed red-tan margins and well- developed phagocytosis of dead cells as well as granulation tissue at the margins []. The LAD branch showed atherosclerotic changes in the wall with remnants of a thrombus attached to the intima (Fig. ).0 |
A 74-year-old male patient with a history of a previously diagnosed myxoinflammatory fibroblastic sarcoma of the left lower extremity treated with definitive surgery presented <2 years later for the development a new tender area of nodularity distal to his previous surgical incision site. The patient's past medical history was also significant for early stage nonsmall cell cancer approximately 20 years prior, which was treated surgically, as well as localized prostate cancer treated with radical prostatectomy.\nThe patient had initially presented with a mass on the right anterior-medial shin, which at the time measured approximately 4.5 cm. He subsequently underwent an excisional biopsy which revealed low-grade myxoinflammatory fibroblastic sarcoma. One month later, the patient underwent a wide local excision and negative margins were achieved. The patient then underwent skin graft placement and close observation with no adjuvant therapy.\nFourteen months later, the patient noted the development of a tender nodule on the anterior/inferior edge of his prior surgical scar. The patient underwent wide local excision with pathological evaluation showing recurrent myxoinflammatory fibroblastic sarcoma measuring 2.5 cm in size, with extension to the deep margin and a close 1 mm anterior margin. A metastatic workup including CT scans of the chest, abdomen, and pelvis revealed no evidence for metastatic progression of the tumor. An MRI scan 1 month following surgery revealed an “enhancing subcutaneous nodule on the lateral lower leg at the proximal surgical margin consistent with residual recurrent tumor” (Fig. ). The tumor showed contact with the underlying superficial fascia of the lateral compartment although no discrete invasion, swelling, or dysfunction was present.\nAfter multidisciplinary tumor board discussion, the decision was made to treat the patient with preoperative radiation therapy, followed by oncologic orthopedic surgery due to the recurrent nature of the patient's disease. The patient was prescribed a course of radiation therapy to a dose of 5000 cGy in 25 fractions with 6-MV photons delivered with an intensity-modulated radiation therapy technique (IMRT) (Fig. ). This was in an effort to spare bone and reduce the risk of lymphedema as the patient had previously undergone multiple surgical procedures. The patient underwent a wide surgical resection and skin grafting procedure approximately 3 weeks after the completion of radiation therapy. The surgical resection encompassed a 2–3 cm margin around the patient's residual disease and included a portion of the anterior compartment, lateral compartment, and small portion of the gastrocnemius muscle posteriorly (Fig. ). Microscopically, the tumor is composed of areas of solid sheets of atypical cells with marked nuclear pleomorphism. There are also areas with myxoid stroma. Scattered bizarre multinucleated giant cells are present. Rare mitotic figures are seen. There are no areas of necrosis (Fig. ). The tumor had focal fascial invasion without underlying muscle involvement, and clear negative margins were achieved. Following wide surgical excision, the patient underwent a complex closure requiring split thickness skin grafting. At 3-month follow-up, the patient continues to recover from surgery, and postoperative imaging shows no evidence of metastatic disease on CT of the chest. MRI of the lower extremity shows interval postsurgical change with no evidence of nodular mass like signal abnormality or enhancement. Clinically, the patient remains neurologically intact in the lower extremity, with little-to-no evidence of lymphedema following surgery. He did experience some loss of his skin graft at the distal aspect requiring vacuum-assisted closure and will soon undergo additional skin grafting to correct this defect. |
The patient was an 80 year old man admitted to the ER with abdominal pain. At admittance, the patient was unable to walk and described an increased difficulty with movement and coordination of the lower limbs, tendency to fall, extensive fatigue, and pain. Six months earlier, the patient had full movement of his lower limbs and could walk unaided. He described an increased intensity and duration of symptoms during the last three months. He was also confused and was unable to answer correctly on orienting questions. Blood serum samples taken at the ER showed a slight thrombocytopenia and mild kidney insufficiency. An arterial blood gas showed hypercalcemia. His condition was deemed to be a combination of somatic pain and confusion. He was therefore treated as a mild trauma patient. Computer tomography (CT) of the abdomen was performed and a surgical consult ruled out any surgical pathology. A pelvis CT showed deterioration of the right hip and an orthopedic consult diagnosed right-sided hip arthritis. The patient was admitted to the orthopedic trauma ward for one day before being transferred to the geriatric clinic for mobilization and pain optimization. He was placed at the neurogeriatric ward to due to space constraints caused by Covid-19.\nAt the neurogeriatric ward, clinical examination revealed several neurological abnormalities including apraxia, dysdiadochokinesia, intentional tremor, clonus, and parkinsonism. Both sides of his body were affected although the left side showed more pronounced symptoms than the right. He was paraspastic and there was parkinsonism in his upper extremities with cogwheel rigidity bilaterally. There was hyporeactivity in his triceps, biceps and brachioradialis tendon reflexes. His lower body display mixed neurological symptoms with peripheral rigidity and hyporeactivity in quadriceps tendons reflexes. He admitted no pain sensation in his left lower leg and foot, though both proprioception and sense of vibration were preserved. He had positive Babinskis sign bilaterally, indicative of damage to the pyramidal neuronal pathways as well as Glabellar tap sign indicating a primitive frontal release, concurrent with parkinsonism. He had difficulty with coordination and was unable to perform fine motor skill tests using his left hand. He was only capable of a wide gait and unable to performed quick turns or adjust walking speed even while using walking aid. He had orthostatic hypotension and was prone to falling. His Mini Mental State Examination score was 9/30 and he had problems with short-term memory, orientation and recall (Table ).\nAnamnesis from the patient and his next of kin revealed that he had been suffering from symptoms such as diarrhea, urine retention, fatigue, and pain for at least six months. The symptoms were infrequent but had become more and more common before admittance to the hospital. He had also lost weight, estimated at 20% of his body weight. His partner noted that he had become more forgetful and occasionally did not recognize acquaintances and family members. He had trouble walking and moving around the house. Sometimes he would appear to be confused about everyday tasks. There was no family history of dementia or cognitive impairment. The patient had worked as a salesperson up until retirement at age 67 and had up until six months before admittance to the hospital not shown any signs of memory impairment or dementia. His deterioration, by all accounts, had been sudden and quick. He was subsequently treated as a patient with suspected cognitive impairment of an unknown cause and standard examinations and tests followed.\nA brain CT revealed white matter changes corresponding to Fazekas stage 2 and medial temporal atrophy grade 2–3. The hippocampus was significantly reduced in volume bilaterally (Fig. ). There was a light global atrophy and no signs of increased intracranial pressure. There was a lytic destruction in the left frontal bone, indicative of a metastasis (Fig. ).\nApoE analysis showed E3/E3 variant. Electroencephalography that showed marked abnormality with laterality to the left hemisphere and loss of alpha rhythm. Extended blood panel showed highly elevated serum calcium and lactate dehydrogenase.\nCSF analysis showed increased albumin at 1230 mg/L (ref < 400 mg/L), severely reduced Amyloid beta (Aβ) 42 at 265 ng/L but with normal amyloid ratio at 0,9 (ref > 0,59), elevated neurofilament light protein at 4600 ng/L. Tau protein as well as phosphorylated Tau protein were within normal range values. Immunoglobulin assay showed increased levels. Paraneoplastic antibodies were negative.\nA diagnosis of possible AD was confirmed using findings of degenerative changes on brain CT, clinical status, and pathological electroencephalography. However, CSF amyloid biomarkers findings did not correlate to AD. Furthermore, neither the full spectrum of his neurological symptoms nor the very high CSF albumin levels could be explained by an AD diagnosis. These findings, coupled with the lytic destructive lesion in the frontal bone and pronounced paraspasticity, prompted further examination.\nA spinal MRI revealed a large lytic tumor incapsulating the whole of Th8 as well as parts of Th7 and Th9 (Fig. ). Metastasis were found throughout the spine and the pelvis. Biopsy and immunocytochemistry confirmed the tumor to be a plasmacytoma containing both necrotic tissue and abnormal plasma cells. The primary tumor compressed the medulla spinalis and myelopathy was present within the spinal cord in association with the tumor.\nThe patient received oncological and hematological consultations and was then started on high dose Prednisolone, Bortezomib and radiation therapy. Neurosurgical intervention was dismissed considering the large involvement of the tumor and the already significant tissue damage. The patient was kept at a geriatric emergency ward and received both medical treatment as well as daily rehabilitation and physical therapy.\nTreatment enabled some reduction in tumor size and after one month the patient experienced slight improvement with absence of tremor and improved walking ability. His rigidity was also less substantial, and he experienced less fatigue. Although his cognitive abilities were not improved, neither had they deteriorated any further. His prognosis at this stage was considered good given his response to treatment. |
A 44-year-old Moroccan Jew woman (gravida 4, para 4) was admitted to an internal medicine ward for general weakness and drowsiness, which began 2 days before admission. Her past medical history included morbid obesity and related complications of type 2 diabetes, which were controlled by metformin treatment; dyslipidemia, which was treated with atorvastatin; gastroesophageal reflux disease, which was not being managed with regular treatment; and fatty liver disease with no other known comorbidities. Her family history included untimely deaths of two of her four children, whose symptoms were similar to those in Leigh’s disease; her other two children are alive and healthy. Aiming to lose weight and control additional comorbidities, she underwent gastric band surgery at the age of 30 years. At the age of 38 years, owing to insufficient weight loss, she underwent gastric sleeve bariatric surgery, which resulted in a weight loss of 30 kg; however, she rapidly regained the lost weight. She subsequently remained morbidly obese, with a body mass index of 48.4. She was approved for a third bariatric surgery involving gastric bypass, and was admitted at the age of 44 years, weighing 126 kg. The surgery began with a laparoscopic approach, but, because of bleeding and gastrointestinal leakage, the procedure was converted to an open laparotomy. The procedure included an omega loop gastric bypass and an elective gallbladder resection, but was complicated by heavy postoperative bleeding. During the next several months, she underwent prolonged hospitalization with continuous mechanical ventilation, which required insertion of a tracheostomy tube, several recurring surgeries for peritoneal lavage, and repeated abscess drainage from the internal abdomen and abdominal wall. During this period, the patient also experienced recurrent episodes of sepsis, which required treatment with multiple courses of broad-spectrum antibiotics. Eventually, after 8 months of postoperative hospitalization, she recovered and was transferred to the rehabilitation department.\nAfter 44 days in the rehabilitation department, she gradually developed new symptoms including slow psychomotor response, weakness, worsening peripheral edema, and drowsiness. Her vital signs on admission to the internal medicine department were unremarkable: temperature 36.4 °C, blood pressure 110/72 mmHg, pulse rate 84 beats per minute, respiratory rate 22 breaths per minute, and oxygen saturation on room air 97%. Her weight was 95 kg. Physical examination revealed grade 3 encephalopathy with asterixis, and neurological evaluation revealed hyporeflexia with symmetric weakness and decreased muscle tone. Other findings included peripheral edema grade 3. The surgical wounds in the abdominal wall had closed, and no signs of infection were seen. No tenderness was observed during abdominal palpation. Laboratory blood examinations showed an elevated international normalized ratio (1.6; normal range: below 1.1), severe hypoalbuminemia (1.5 g/dL; normal range: 3.4–5.4 g/dL), and highly elevated blood ammonia (285 µg/dL; normal range: 15–45 µg/dL). Other results included hemoglobin 9.2 g/L, white blood cell count 7200/µL, platelets 234,000/µL, glucose 91 mg/dL, urea 33 mg/dL, creatinine 0.7 mg/dL, sodium 137 mmol/L, potassium 4.3 mmol/L, magnesium 2.4 mmol/L, calcium 7.2 mmol/L, phosphorus 3.4 mmol/L, aspartate aminotransferase (AST) 28 IU/L, alanine aminotransferase (ALT) 31 IU/L, alkaline phosphatase 131 IU/L, gamma glutamyl transpeptidase 34 IU/L, lactate dehydrogenase 413 IU/L, total bilirubin 0.9 mg/dL, creatine phosphokinase 89 IU/L, pH 7.34, bicarbonate 23 mg/dL and PCO2 39.6 mmHg. Her C-reactive protein level was 1.2 mg/L (normal: 0–0.5 mg/L), and her blood ketones were negative. Further laboratory tests revealed normal levels of factor 5 and normal lactate levels (0.6 mg/dL). Imaging studies, including abdominal ultrasound and computerized tomography (CT), did not reveal any signs of cirrhosis; apart from previously known postsurgical changes, only diffuse fatty infiltrates of the liver were found (Fig. ). Brain CT, including venography and angiography, showed no signs of intracranial pathology, ischemia, or early or late enhancement. Her electroencephalographic findings supported the diagnosis of encephalopathy.\nThe most likely diagnosis was urea cycle disorder (UCD), as supported by the high ammonia levels, low blood protein levels, and advanced encephalopathy. Imaging studies showed no signs of cirrhosis, and laboratory investigations excluded hepatocellular injury. Hepatic cellular function was preserved, as reflected by standard factor 5 levels. Plasma amino acid analysis revealed a decrease in levels of several amino acid: taurine 17 µM, aspartate 13 µM, citrulline 4 µM, valine 105 µM, leucine 14 µM, tyrosine 25 µM, and histidine 58 µM. Other amino acids were in normal ranges. An acylcarnitine profile showed elevated carnitine with no further abnormalities. The urine organic acid profile revealed markedly elevated 4-hydroxyphenyllactate, but this test was inconclusive because the high levels of antibiotics in the blood might have influenced the results. Karyotyping revealed no irregularities.\nEmpirical treatments with the ammonia scavenger sodium benzoate, N-carbamylglutamate, l-arginine, carnitine, and low-protein total parenteral nutrition were administered in addition to nutritional supplements, which included thiamine, zinc, and vitamins C, B, and E. The patient’s encephalopathy condition continued to deteriorate, with new-onset fever and hemodynamic instability combined with respiratory failure. She was placed on mechanical ventilation, supported by vasopressors, started on a course of broad-spectrum antibiotics, and transferred to the internal intensive care unit, where a second ammonia level test was conducted and indicated a marked increase to 455 µg/dL. Her high ammonia levels were controlled by hemofiltration, in addition to the above treatment, and by administration of supplemental intravenous amino acids. Hemofiltration helped control her ammonia levels, which gradually decreased to baseline levels within 5 days.\nA sepsis investigation began after fever and hemodynamic instability further complicated her condition, but no infectious source was found, and all cultures were negative. Total body CT and cardiac echocardiography were performed and yielded normal results.\nWith the resolution of systemic inflammatory response syndrome (SIRS), our patient was gradually weaned from mechanical ventilation and vasopressors, and her nutrition was steadily shifted from parenteral nutrition to enteral nutrition, and later to oral nutrition. The patient recovered neurologically and regained normal cognitive function. She subsequently developed critical care polyneuropathy, owing to the use of muscle relaxants, and was discharged to undergo rehabilitation in a designated hospital without a further need for nutritional supplements.\nAfter 2 months in the rehabilitation department, she was discharged home. At a 2-year follow up after the hospitalization described above, her hyperammonemia had not recurred, and she had recovered from critical care polyneuropathy. During the follow-up period, no liver disease was found, and her ammonia levels remained stable and in the normal range, as did all other examined blood measurements. |
An 86-year-old female patient (145 cm, 35 kg) visited our hospital due to worsening dyspnea. Ten years prior, she was diagnosed with papillary thyroid cancer with invasion of the tracheal tissue, which was treated palliatively with a total thyroidectomy and radioactive iodine (RAI) therapy. Despite the RAI therapy, the thyroid cancer remained in the trachea, and the patient required further supportive treatment.\nSix months prior, she was admitted to our hospital and underwent endotracheal intubation and mechanical ventilation due to respiratory failure. Computed tomography (CT) following intubation revealed an endotracheal mass filling the tracheal lumen. We recommended a palliative tracheostomy to wean the patient off the ventilator, but her family refused this procedure. Therefore, we decided to extubate and perform an endoscopic de-bulking procedure. During the bronchoscope-guided extubation, we found that the endotracheal mass was sufficiently compressed so as not to interfere with airway patency. Thus, we did not perform the bronchoscopic de-bulking intervention, and she was discharged with improvement.\nTo evaluate her current tracheal stenosis, a CT scan was performed. The results showed near-complete obstruction of the upper trachea by a cancerous invasion (). We contacted the interventional radiologist to insert a tracheal stent under balloon dilatation, but he refused to perform the procedure due to the proximity of the mass to the vocal cords. We then recommended two procedures to the patient: a rigid bronchoscopy or a palliative tracheostomy under general anesthesia. The patient refused both recommended procedures, and a subsequent recommendation was made for an intervention employing a flexible bronchoscope. After the patient provided informed consent pertaining to the risks associated with a flexible bronchoscopic procedure, including respiratory arrest, electrical burn, and bleeding, intervention with the flexible bronchoscope was attempted to relieve her tracheal obstruction after injecting 3 mg of midazolam.\nThe patient's upper trachea showed luminal narrowing of more than 75% of the diameter, 1.5 cm distal to the vocal cords, due to a polypoid mass with a soft consistency (). We attempted electrical snaring to reduce the cancerous mass in the trachea, but this procedure was not successful due to severe coughing. Next, we inserted an endotracheal tube with an 8.0-mm inner diameter through the narrowed lumen under bronchoscopic guidance. The cuff balloon, which was located in the narrowest part of the lumen, was maximally inflated for 15 seconds, after which the endotracheal tube was removed. Upon repeated flexible bronchoscopic examination, the endotracheal mass was found to be compressed (), and the patient's cough had decreased sufficiently to perform electrical snaring and argon plasma coagulation of the tumor. Immediately after the procedure, her dyspnea improved markedly (). Six hours after completion of treatment, flexible laryngoscopy was performed to evaluate the mobility of the vocal cords, and no immediate complications were noted. After 4 days of hospitalization, she was discharged. Tracheal patency has been maintained for 8 months. |
Case II. A 40-year-old woman with two early pregnancy losses is found to have persistent presence of lupus anticoagulant. Should she be counseled for antithrombotic treatment to prevent a third pregnancy loss?\nObstetrical complications of the antiphospholipid syndrome can manifest in women with and without a history of thrombotic events. These include recurrent early pregnancy loss, fetal death or (pre)eclampsia, intrauterine growth restriction, and other consequences of placental insufficiency. Traditionally it is hypothesized that pregnancy complications in antiphospholipid syndrome are the result of a hypercoagulable state, partially mediated by thrombosis of the placental vasculature. Recent hypotheses describe a more intertwined pathophysiological mechanism in which the coagulation system as well as inflammation are involved., , , The inhibitory effect of antiphospholipid antibodies on proliferation of trophoblasts of the placenta has been proposed as the pathogenic mechanism in early pregnancy loss, whereas late obstetrical complications have been attributed to a dysfunctional vasculature of the placenta., , , These placenta-mediated complications include preeclampsia, late pregnancy loss, placental abruption, and intrauterine growth restriction.\nPossible effects on complement activation may be of more importance and it has been hypothesized that the non-anticoagulant effects of heparins on inflammatory processes, vascular function, or placental pathology may play a role in prevention of pre-eclampsia, a disorder strongly associated with antiphospholipid syndrome., Moreover, antiphospholipid antibodies appear to affect the production of several chemokines and angiogenic factors by human endometrial endothelial cells, which may contribute to impaired placentation and vascular transformation. The risk of (recurrent) pregnancy complications may differ between women with and without previous complications, women with high and low antiphospholipid antibodies titers, and women with positive and negative LAC., , Antithrombotic therapy reduces the risk of recurrent (either venous or arterial) thrombosis in antiphospholipid syndrome., Both aspirin and heparin may have a beneficial effect on coagulation and inflammation,, , and are thought to reduce the risk of pregnancy loss in antiphospholipid syndrome.\nTo answer the questions posed by our patients, we performed a systematic review and meta-analyses of the evidence available from randomized trials to evaluate the effects of different antithrombotic therapies on pregnancy outcome in women with recurrent pregnancy loss and antiphospholipid antibodies., As antiphospholipid syndrome is a heterogeneous disease, we chose to focus specifically on women with a history of recurrent pregnancy loss. The primary outcome was defined as live birth. Eleven trials including 1672 women met the inclusion criteria. None of the trials had a no treatment comparator arm. Full details of the methods and extracted data are described in the supporting information. Here, we summarize our findings by addressing the questions most important for clinical practice, “who, what, and how” (Table ).\nBased on the individual history of obstetrical complications, treatment during the subsequent pregnancy can be considered. Table provides an overview of current guidelines and recommendations for preventing pregnancy loss in women with antiphospholipid syndrome, stratified for history of obstetrical complications. It is important to note that all available evidence underlying these recommendations concerns women with persistent antiphospholipid antibodies and recurrent early pregnancy loss. High-level evidence for the other clinical criteria is virtually absent and management suggestions are extrapolated from mostly observational evidence and expert opinion. Non-criteria obstetric antiphospholipid syndrome is defined as two early pregnancy losses or delivery after 34 weeks of gestation due to severe (pre)eclampsia. In these women treatment might be considered based on the individual’s risk profile, for instance a high-risk antiphospholipid antibody profile but no history of thrombosis or pregnancy complications. A high-risk antiphospholipid antibody profile is defined as presence of lupus anticoagulant, double or triple antiphospholipid antibody positivity, or persistently high antiphospholipid antibody titers.\nOur search identified 11 randomized trials evaluating antithrombotic treatment in women with recurrent pregnancy loss and antiphospholipid syndrome. Study characteristics are presented in Table . The identified trials differed in terms of inclusion criteria and compared a variety of interventions. One trial compared aspirin with placebo, five trials compared heparin (unfractionated heparin [UFH] or LMWH) plus aspirin with aspirin only,, , , , one trial compared LMWH with aspirin, two trials compared LMWH with UFH (both in combination with aspirin),, and two trials investigated the combination of different doses of heparin (either UFH or LMWH) with aspirin., We did not identify trials with a no treatment comparator arm during pregnancy. Three of 11 trials included women with two or more pregnancy losses. In 8 of 11 trials participants met the clinical criteria for antiphospholipid syndrome with three or more early miscarriages. The mean number of previous pregnancy losses ranged from 3.0 to 4.3. Previous pregnancy losses were mostly early pregnancy losses, but this was only specified in 5 of 11 included studies. All trials included participants with persistent presence of antiphospholipid antibodies, but the timeframe between tests varied.\nThe use of aspirin during pregnancy in antiphospholipid syndrome is widespread. Our search identified one placebo-controlled trial of 40 women with antiphospholipid antibodies and recurrent pregnancy loss evaluating aspirin treatment. This trial, at high risk of attrition bias due to incomplete reporting of outcome data, found no difference in live birth rate with aspirin compared to placebo (risk ratio [RR] 0.94; 95% confidence interval [CI] 0.71–1.25; GRADE very low-certainty evidence, Figure ).\nThe small sample size and methodological limitations hamper the conclusions that can be drawn from this study and these results do not provide evidence to support aspirin only for prevention of pregnancy loss in this population. In the general population as well as in women with a history of one to two previous pregnancy losses, preconception aspirin does not increase live births, as shown in the EAGER trial. However, aspirin is effective in reducing the risk of preeclampsia in high-risk women., Therefore, considering antiphospholipid antibodies a risk factor for preeclampsia, it is very reasonable to use aspirin for prevention of preeclampsia in women with recurrent pregnancy loss and antiphospholipid syndrome.\nOne trial of 141 women with antiphospholipid syndrome reported the results of a head-to-head comparison of LMWH only and aspirin only. Women treated with LMWH had a higher live birth rate of 86.3%, compared to a 72.1% live birth rate in the women treated with aspirin only (RR 1.20, 95% CI 1.00–1.43, 1 trial, 141 women, Figure in supporting information). All other trials evaluated heparin in combination with aspirin.\nFive trials with a total of 1295 women that compared heparin (either UFH or LMWH) combined with aspirin to aspirin only, were included in a random-effects meta-analysis for the primary outcome live birth. The pooled RR for live birth was 1.27 (95% CI 1.09–1.49; Tau2 = 0.01; Chi2 = 7.71, I2 = 48%; GRADE low-certainty evidence) in favor of heparin plus aspirin compared to aspirin only., , , , There was significant heterogeneity between the subgroups of LMWH and UFH (RR for LWMH plus aspirin versus aspirin 1.20, 95% CI: 1.04–1.38; RR for UFH plus aspirin versus aspirin 1.74, 95% CI: 1.28–2.35; test for subgroup differences: I2 =78.9%, p = .03, Figure ). The observed live birth rate in the aspirin-only comparator arms of the UFH studies was considerably lower compared to these in the LMWH studies; 42.9% versus 70.4%. We performed a sensitivity analysis excluding two studies for serious methodological limitations; one (n = 50) at high risk of selection bias due to the quasi-randomized design, one (n = 1015) at high risk of attrition bias due to incomplete reporting of outcome data. This did not materially change the combined (UFH +LMWH) pooled result (RR for live birth 1.20, 95% CI 0.91–1.59; I2 = 58%), but the benefit of LMWH plus aspirin compared to aspirin only was attenuated (RR for live birth 1.07; 95% CI 0.88–1.29, Figure ). Furthermore, after excluding the largest and most recent trial, the statistical significance for all heparin trials was lost.\nThere was no statistically significant difference in live birth between LMWH and aspirin versus UFH and aspirin (RR 1.44, 95% CI 0.80–2.62, 2 trials, 86 women; p = .17; I2 = 48%; Figure in supporting information)., Heparin appears to improve live birth rates, but the low live birth rates in the comparator arms in the UFH studies may lead to an overestimation of the effect of UFH. The observed beneficial effect of LMWH plus aspirin on the other hand is mostly driven by a recently published large single-center trial (n = 1015) that found a 90.3% live birth rate in women treated with LMWH plus aspirin, compared to 70.1% in those treated with aspirin only. Table provides an overview of study outcomes and certainty of the evidence for the two main comparisons: (1) aspirin versus placebo and (2) heparin plus aspirin versus aspirin only.\nNotably, adverse events associated with heparin therapy, easy bruising at injection site or allergies, did not seem to occur frequently or were not reported. LMWH is a reasonable alternative treatment and currently most often used in clinical practice, with its similar efficacy and superior safety profile compared to UFH.\nWe observed a wide variation in treatment initiation and duration between trials. Aspirin treatment was started preconceptionally in most trials and continued to 36 weeks of gestation, , , or full-term pregnancy., LMWH or UFH was started upon pregnancy confirmation in most studies evaluating heparin treatment. In the trial by Rai et al., aspirin treatment was started upon pregnancy confirmation and when fetal heart activity was confirmed on ultrasound women were randomized to additionally start UFH or continue aspirin only. Four trials initiated treatment at the first confirmation of pregnancy and treatment was continued until 34 weeks of gestation, 35 weeks of gestation,, or study duration. One trial started aspirin before conception, with heparin (LMWH or UFH) started in the luteal phase for a maximum of three cycles until delivery and continued postpartum in a prophylactic dose. The mean gestational age at randomization, and thus treatment initiation, varied largely between studies with one study allowing randomization before 12 weeks of gestation. Given the heterogeneity in treatment protocols, it is not possible to provide recommendations on optimal timing of treatment initiation and duration. A recent study that compared early initiation of LMWH (gestational age 5 weeks) to later initiation observed ongoing pregnancy rates of 81% at 12 weeks’ gestation and 61%, respectively. Live birth rates differed between the groups, 70.8% in the early initiation group and 56.5% in the later initiation group, respectively, but this difference was not statistically significant. Also, late pregnancy complications associated with antiphospholipid syndrome, including preeclampsia, intrauterine growth restriction, and intrauterine fetal death, were not statistically significantly different between the two study groups. Similarly, another placebo-controlled trial reported higher ongoing pregnancy rates at 24 weeks’ gestation in women treated with LMWH and aspirin preconceptionally, but live birth overall was not affected. Initiation of heparin preconceptionally would be undesirable from a patient’s perspective, but whether heparin can be safely discontinued after the first trimester of pregnancy requires further investigation. Three studies continued heparin treatment postpartum; either 3 weeks, or 6 weeks. The incidence of postpartum thrombosis in women with obstetric antiphospholipid syndrome is unknown. Therefore, the aim and duration of postpartum heparin treatment cannot be evidence based. In the absence of evidence, however, women with persistent antiphospholipid antibodies may be at higher risk of thrombosis and postpartum continuation of heparin treatment for prevention of venous thromboembolism can be considered.\nVarious doses of aspirin and heparin were used in the included studies (Table ). Due to small study sample sizes and limited data it was not possible to account for these differences in the analyses. Two small trials compared a higher and a lower dose of heparin (LMWH or UFH) both combined with aspirin.\nA higher dose of LMWH did not improve the live birth rate compared to a lower dose of LMWH (RR 1.10, 95% CI 0.81 to 1.49, 1 trial, 60 women); similar to the effects of a higher dose of UFH compared to a lower dose of UFH (RR 1.05, 95% CI 0.78 to 1.41, 1 trial, 50 women; Figure in supporting information)., Importantly, the study evaluating different doses of UFH lacked the power to detect any significant differences and had methodological limitations due to the quasi-randomized design. This variation in initiation of treatment, in duration of treatment, as well as different doses and agents used, limits the possibilities of a cross-study comparison and thus clear treatment recommendations.\nIn general, although we focused on women with recurrent pregnancy loss, we observed significant clinical heterogeneity in the study populations. A substantial part of the studied population did not meet the full criteria of antiphospholipid syndrome, due to differences in the definition of prior pregnancy loss used as well as the timing (and interval) of repeat antibody testing. Criteria for antiphospholipid syndrome and pregnancy loss are consensus based and further research regarding which subgroups benefit from antithrombotic therapy should be carried out. It is known that the prognosis varies between subgroups of antiphospholipid syndrome patients., , , , , Reporting of antibody profiles or the number of previous pregnancy losses was incomplete and not in relation to the primary outcome live birth. For this reason, we were unable to perform subgroup comparisons based on number of previous miscarriages (two vs. three or more), previous placenta-mediated complications, high-titer antibodies versus low-titer antibodies, or positive LAC versus negative LAC.\nIn light of the limitations of the included studies in this review, the evidence is of low certainty, and a large multicenter randomized controlled trial with clearly defined patients with antiphospholipid antibodies and recurrent pregnancy loss is still warranted. Such a trial should include women with homogenous clinical characteristics and antibody profiles or be powered to analyze subgroups. We realize this is challenging but given the costs, nuisance, and side effects of heparin and aspirin observed in trials in non-antiphospholipid syndrome patients,, such trials should be performed to obtain a definite answer about the effectiveness in antiphospholipid syndrome. Unfortunately, despite the urgent need to get answers to our important clinical questions, this is challenging. For instance, the well-designed APPLE pilot study evaluating LMWH plus aspirin versus aspirin only during pregnancy in women with persistent antiphospholipid antibodies and a history of two or more early pregnancy losses or one or more late losses was terminated prematurely for feasibility reasons.\nOur systematic review provides a contemporary and complete synthesis of all available evidence from randomized trials on antithrombotic therapy for improving pregnancy outcomes in women with a history of recurrent pregnancy loss and persistent presence of antiphospholipid antibodies. Based on the available but low-certainty evidence, heparin plus aspirin appear to improve live birth rates in women with recurrent pregnancy loss and persistent antiphospholipid antibodies.\nSo how do these findings translate to our own clinical practice? To summarize our “who, what, and how” for women with antiphospholipid antibodies and recurrent pregnancy loss:\nWho do we treat? Women with recurrent early pregnancy loss (three or more) and persistent presence of antiphospholipid antibodies tested on two separate occasions at least 12 weeks apart. In women with a late pregnancy loss or late pregnancy complications in persistent presence of antiphospholipid antibodies treatment, treatment will be discussed as we also counsel these women on the absence of evidence on its effectiveness.\nWhat do we prescribe? A combination of low-dose aspirin and LMWH in prophylactic dose.\nHow do we treat? Aspirin is started preconceptionally with LMWH added upon pregnancy confirmation. Treatment is continued for the full duration of pregnancy and stopped at either the first signs of labor or 24 hours prior to planned delivery. We consider continuation of LMWH postpartum based on the individual patient's risk profile for venous thromboembolism. |
A 19-year-old female patient presented to our University Dental Center with the chief complaint of deficient maxillary anterior edentulous ridge. The patient wanted the esthetic rehabilitation of missing upper front teeth. On clinical examination, there was deficient maxillary anterior edentulous ridge complicated by the traumatic deep bite. After taking initial records [], the case was discussed among prosthodontist, periodontist, and orthodontist. After the oral prophylaxis and hygiene maintenance instructions patient was referred to the orthodontist for bite opening. It took 12 months to open the bite []. The patient was then referred to the periodontist for the management of deficient maxillary anterior edentulous ridge. A consultation with prosthodontist involving patient was made before ridge splitting procedure regarding the rehabilitation of edentulous maxillary anterior segment using dental implants or fixed prosthetic bridge. Due to economic conditions patient opted for a fixed bridge in place of dental implants. For achieving an emergence profile of the prosthesis, improvement in the bone height and width was required. Hence, it was decided to perform ridge splitting procedure for improving horizontal and vertical thickness of ridge using bone allograft without implant placement at the time of ridge splitting procedure.\nIt is not extremely common to open an edentulous ridge, only to find thin atrophic bone especially when it is traumatized by opposing teeth. In this case, a SRSP was performed to open entire edentulous bony segment like an envelope to receive the bone graft. After routine preparation of the surgical site with a Povidine Iodine solution (5% w/v), the local anesthetic solution (2% adrenaline) was administered. The incision was made with no. 15 Bard Parker blade at midcrestal level and the full thickness mucoperiosteal flap was raised. On reflection of mucoperiosteal flap, it was found that the palatal cortical bone was mostly absent due to traumatic deep bite from mandibular anterior teeth. The sharp bony spicules were rounded off by osteoplasty procedure to provide a smooth blended bone surface. The edentulous alveolar ridge was then splitted vertically by inserting chisel. The chisel was directed towards the palatal side to give the access for the placement and retention of bone graft in the ridge. The decalcified freeze dried bone allograft (DFDBA) was prepared by processing the graft particles into a sterile dappen dish and reconstituted with sterile saline water. Excess sterile saline was removed with a gauge; the graft material was then placed into the surgically prepared site with an amalgam carrier followed by firm pressure with amalgam condenser. The defect was filled to the existing alveolar crest. Primary soft tissue closer was done using 3–0 black braided silk suture by simple interrupted sutures. Periodontal dressing was given for the wound stabilization for 7 days [].\nPostoperatively, the patient was placed on amoxicillin 250 mg and metronidazole 200 mg 8 hourly for 5 days. Ibuprofen 400 mg 8 hourly for 3 days and chlorhexidine gluconate mouth rinse (0.12%) twice daily 10 ml for 2 weeks. The patient was instructed neither to brush the surgical site nor manipulate it in any manner for 10 days. The sutures were removed after a week and the site was debrided with saline water. Splinted porcelain fused to metal bridge was then delivered []. We have followed the case for 6 months. |
A 69-year-old Asian female presented to a hospital with moderate chest pain that began 3 weeks prior to surgery. She was initially evaluated and managed with antihypertensive and pain medications. However, when the symptoms reoccurred as a new episode of crushing and sharp midthoracic back pain, the patient presented to a different emergency department and was transferred to the Johns Hopkins Hospital. Clinical workup included a chest computed tomography (CT) angiogram that characterized an aorta with diffuse mural thickening. The hematoma was circumferential in nature and measured 8 mm in thickness at the level of the aortic root throughout the ascending aorta, and grew to 15 mm in the descending aorta (\n). The ascending aorta was also mildly dilated and measured 37 mm in diameter at the level of the pulmonary artery bifurcation. Transthoracic echocardiography (TTE) demonstrated well-preserved left ventricular function with an ejection fraction estimated at 60%, without regional wall motion or valvular abnormalities. Cardiac CT scan with infusion of the coronary arteries suggested the presence of moderate coronary artery disease with no hemodynamically significant stenosis. Based on these findings, a diagnosis of IMH was confirmed, and the patient was advised to undergo urgent surgical repair.\nA median sternotomy was performed, and once the pericardium was opened, a moderate bloody effusion was drained. The ascending aorta was visibly affected by an extensive hematoma presenting with bluish discoloration of the adventitia (\n). The patient was cannulated with the Seldinger technique directly into the distal ascending aorta under TEE guidance to confirm placement into the true aortic lumen. Venous cannulation was bicaval in the superior and inferior vena cava. Deep hypothermic circulatory arrest was performed at 18°C and maintained for 41 minutes. Cerebral protection was performed with retrograde cerebral perfusion at 300 mL/min. Once on cardiopulmonary bypass and following institution of deep hypothermic cardiocirculatory arrest, the proximal ascending aorta was noted to contain a periaortic IMH upon incision. The IMH extended circumferentially through the entire length of the ascending aorta from the noncoronary sinus of the aortic root to the abdominal aorta, dissecting the medial and intimal layers of the vessel (\n). Inspection of the aortic arch for areas of dissection was performed and demonstrated no additional intimal injury, intimal flaps, or entry sites. Therefore, ascending aorta was replaced entirely to the hemiarch with a 30-mm Hemashield platinum graft. The aortic root layers were recompacted within a sandwich of Teflon strips. Postrepair, intraoperative TEE verified the integrity of the aortic repair with no changes in the aortic valve's preserved continence. The patient's postoperative course was unremarkable, and she was discharged home on postoperative day 5. The descending aorta will be monitored with yearly CT angiography.\nPostoperative pathology findings revealed cystic medial degeneration of the aortic wall, characterized by elastic fiber fragmentation with the formation of mucopolysaccharide pools and medionecrosis, defined as an apparent loss of nuclei in the media (\n). Blood was also noted to split the medial and intimal layers of the aorta (\n). |
A 73-year-old man with multiple medical comorbidities including multiple myeloma after chemotherapy, diabetes, and coronary artery disease presented for the treatment of a comminuted right periprosthetic supracondylar distal femur fracture after sustaining a mechanical ground level fall (Figure ). Before this injury, he was able to ambulate two blocks without difficulty but did use a stair lift at home secondary to chronic left lower extremity weakness, which he attributed to his previous multiple myeloma treatment. He was medically optimized and taken to the operating room the following day for open reduction and internal fixation (Figure ). After achieving adequate reduction of the fracture through indirect reduction techniques, a limited lateral approach to the distal femur was performed and the proximal end of a titanium alloy NCB Periprosthetic Plate (Zimmer Biomet) was placed submuscularly. Three titanium alloy 5.0 mm standard screws were placed in a percutaneous manner proximal to the fracture and multiple titanium alloy 5.0 mm locked screws were placed in the distal segment. Postoperatively, he was placed in a knee immobilizer to be worn at all times for 3 weeks, at which time the range of motion exercises were initiated. He was limited to toe-touch weight bearing for 8 weeks. Before discharge, he was evaluated by our endocrinology team who obtained laboratory test results including comprehensive metabolic panel, vitamin D 25 hydroxy, and inorganic phosphate. Several of these laboratory values were found to be abnormal, and he was ultimately started on calcium and vitamin D supplements.\nThe patient was discharged to a skilled nursing facility on postoperative day 5. At his 8-week postoperative appointment, new radiographs were obtained (Figure ). Adequate callous formation was noted, and no concerns for complications were noted. At that time, he did have notable quadriceps atrophy but denied pain with passive or active range of motion. He was advanced to weight bearing as tolerated and given home quadriceps strengthening exercises.\nAt his 14-week postoperative appointment, it was noted that the patient had not been compliant with his home strengthening exercises, and he reported 75% weight bearing with continued use of a walker for all ambulation. He endorsed stiffness in his right knee and intermittent mild pain described as muscle soreness in his distal thigh. Radiographs were obtained, which demonstrated slight varus malalignment of the fracture and fretting of the distal-most screw placed proximal to the fracture site (Figure ). He was referred to physical therapy but was unfortunately lost to further follow-up. |
A 14-year-old boy, who was initially examined for continuous coughing, was referred to our hospital owing to an anterior mediastinal mass identified on chest X-ray (). Systematic examination revealed a 20 × 10 cm sized mass at the right anterior mediastinum that involved the right pulmonary vein and elevated levels of alpha fetoprotein (AFP), a tumor marker, at 3825 ng/ml. Needle aspiration was performed, but only necrotic tissue could be collected. We did not analyze the karyotype since the symptoms suggesting the Klinefelter syndrome were not observed. Therefore, a clinical diagnosis of malignant teratoma was made, and three courses of cisplatin-based chemotherapy were administered because AFP levels continuously increased. After chemotherapy, AFP levels decreased, although the size of the tumor did not change, as evident in a computed tomography (CT) scan. Next, tumorectomy and total right lung extraction were performed. The pathological diagnosis of the extracted tumor was malignant teratoma with areas of yolk sac tumor (). No cancer cells were found at the edges of the area where the tumor was removed. Two courses of cisplatin-based postoperative chemotherapy were administered until AFP levels were normal.\nAfter 3 months of follow-up, the patient experienced pain in his right hip joint while AFP was still normal. The 99mTc-methylene diphosphonate bone scan showed increased tracer uptake at the left forehead and right hip joint (). Head and pelvic MRI also revealed signs of metastasis at the left forehead and right hip joint. Abdominal CT showed a metastatic region in the liver. Bone biopsy was performed at the left forehead. The pathological diagnosis was metastatic malignant melanoma originating from an immature teratoma of mediastinum (). Large heteromorphic cells with melanin were found in the original mediastinal malignant teratoma by retrospective re-examination. This suggested that a section of the malignant melanoma in the original malignant teratoma, which was composed of various components, metastasized. Because of the very fast disease progression, after consultation with the family, aggressive treatment was discontinued, and palliative therapy was provided. He died 15 months after diagnosis of the original malignant teratoma. |
A 36-year-old male patient with a 6-year history of chronic kidney disease of unknown etiology was brought to the emergency department due to neurological impairment that started during the previous hour characterized by altered mental status while he was walking down the street. On examination he was stuporous, with poor response to external stimuli. The patient was admitted to the hospital and vital signs and capillary glucose were determined. Glucose levels were 20 mg/dl and increased to 42 mg/dl after a 50 ml infusion of 50% dextrose. During his stay his mental status recovered after glucose levels were returned to normal parameters, requiring high doses of intravenous glucose. After his stabilization he was transferred to the internal medicine department. We confirmed that the patient was not taking medications that would cause hypoglycemia. On physical examination he was somnolent and pale, with slight oedema in both legs. A new episode of symptomatic hypoglycemia was observed while he was receiving an infusion of 20% dextrose. The infusion rate at that moment was 10.416 ml/hr. The infusion was not being weaned off or was an acute disruption. It caught our attention that although the patient was on a 20% dextrose infusion, he continued with hypoglycemia. Blood samples were taken and the results were abnormal ().\nAlthough hypoglycemia could be explained by chronic kidney disease, the diagnosis of insulinoma was considered, so a computed axial tomography with double contrast was taken but unfortunately there were no abnormal findings. During the patient's hospital stay he received several treatments that failed to achieve proper glucose control. We used ascending doses of diazoxide up to 600 mg/day with a poor response. In order to reduce episodes of hypoglycemia, we started with low doses of octreotide and found a good response that created tolerance quickly, so we decided to increase the dose by 0.1 mcg/kg/h always observing the same phenomenon. We decided to suspend this treatment when we reached 0.4. mcg/kg/h. Finally, we decided to maintain a continuous infusion of 50% dextrose with which we achieved serum glucose levels between 120 and 160 mg/dl. Magnetic resonance imaging and an endoscopic ultrasonography were performed but no conclusive data on any structural pancreatic disorder were obtained. In order to locate the tumor, we performed selective intra-arterial pancreatic stimulation with hepatic venous sampling at 0, 20, 40, and 60 seconds. High insulin levels were obtained after a selective injection of 0.025mEq/Kg calcium gluconate in the proximal splenic and gastroduodenal arteries () (). With these results, the patient was scheduled for surgery. During the procedure, bimanual palpation of the pancreas was performed, as well as a pancreatic ultrasound in which no tumor could be identified. The body and tail of the pancreas were resected. For two days the patient had an adequate glycemic control but after a couple of days, he presented with hypoglycemia again. The macroscopic pathology report did not show any tumor compatible with insulinoma; however, on microscopic examination pancreatic islets with elongated cells and clear cytoplasm compatible with nesidioblastosis were seen (). Unfortunately, during his stay at the intensive care unit, the patient developed late-onset hospital-acquired pneumonia and, in spite of treatment, he developed sepsis followed by septic shock which ultimately caused his death. |
A 39-year-old male presented to our institution with a 3-week history of nausea, vomiting, and diarrhea. Medical history was notable for occipital melanoma excised in 2011. Subsequently during a dental procedure in 2016, the patient was noted to have left sided lymphadenopathy. He underwent a lymph node biopsy, which was positive for metastatic melanoma which resulted in a full neck dissection. Two months thereafter and four weeks prior to his symptom onset, his primary oncologist started him on ipilimumab. The patient then presented to the medical intensive care unit with hypovolemic shock secondary to intractable diarrhea. In addition to fluid resuscitation, patient was continued on his prednisone 50 mg oral twice daily which had been started by his oncologist. Patient's severe diarrhea persisted which prompted endoscopic examination. Colonoscopy showed circumferential, diffuse, edematous, erythematous, and friable mucosa with superficial ulcerations along the entire length of the colon and the terminal ileum (Figures and ). Pathology from the biopsies obtained during the colonoscopy was consistent with chronic active ileocolitis with acute cryptitis, crypt abscess, and granulation tissue (Figures and ). The steroid regimen was consequently changed to intravenous methylprednisolone 20 mg every 8 hours. The patient also underwent a computed tomography enterography which demonstrated narrowing of the distal and terminal ileum as well as a short segment of narrowing in the mid ileum with fecalization in the distal ileum (). Additionally, pericolonic stranding was noted to be prominent in the descending colon. Unfortunately, the patient did not respond to intravenous methylprednisolone over the course of the next 8 days and a decision was made to initiate infliximab at 5 mg/kg. Despite the start of infliximab, the patient continued to worsen with diffuse abdominal pain and persistent diarrhea. A repeat computed tomography of the abdomen and pelvis showed prominent ascending and transverse colitis with pneumatosis of the distal small bowel, perforated abdominal viscus with moderate pneumoperitoneum and pneumomediastinum (). The patient underwent a subtotal colectomy with end ileostomy. The patient's symptoms improved thereafter and almost a month into the hospital admission, the patient was discharged on an oral methylprednisolone taper. |
A 55 year old man was diagnosed with anemia by his family physician towards the end of 2017. In February of 2018, an upper GI endoscopy and colonoscopy were performed which showed a right colon cancer. CEA was 40.4. CT of the chest, abdomen, and pelvis was obtained in February of 2018. This showed a mass 7 cm in greatest diameter that was immediately adjacent to the undersurface of the right liver and contiguous with numerous loops small bowel and the lowest part of the second portion of the duodenum (). There were mildly prominent mesenteric lymph nodes but no evidence of distant metastatic disease.\nIn February of 2018, the patient underwent a single port laparoscopic right colon resection. The surgeon described the tumor as stuck to the undersurface of the liver with adhesions taken down without difficulty. On histopathologic examination, the tumor was PT3N0M0 with 0/16 positive nodes. Perineural invasion was identified. The cancer was moderately to poorly differentiated. Systemic chemotherapy was not recommended.\nIn June of 2018, the patient began noting problems with digestion and pain after eating. CEA had increased to 940 ng/ml. Repeat CT scan showed multiple nodules within the greater omentum compatible with peritoneal metastases. The liver, kidneys, and ureters were normal. There was marked stranding within the right colon resection site (). Bowel loops proximal to the prior ileocolic anastomotic site were moderately dilated and fluid-filled. A mass was noted in the abdominal wall at the laparoscopic port site. There was also a mass in the rectovesical space immediately adjacent or invading the right and left seminal vesicles.\nIn July of 2018, the patient underwent exploratory laparotomy. The posterior surface of the liver was layered by cancer. The undersurface of the right hemidiaphragm was covered by tumor nodules. The pelvis contained a 4 cm tumor mass invading into the seminal vesicles. The old ileocolic anastomosis was involved and needed to be resected. The umbilical port site was infiltrated by cancer. Near complete resection was possible by cytoreductive surgery.\nIntraoperatively, the patient was treated with HIPEC-melphalan []. This postoperative course was unremarkable. The histopathology showed adenocarcinoma compatible with the primary tumor in all tissues submitted. The peritoneal cancer index was 28 and the CC score was 2 []. The resection was judged to be a palliative effort and its major goal was to relieve intestinal obstruction and to prepare the patient for rapid initiation of palliative systemic chemotherapy.\nNeither of our patients was evaluated by the MDT preoperatively. Their preoperative and intraoperative management was evaluated in an attempt to improve the outcome of subsequent patients. |
A 31-year-old gravida 4 para 0 African American woman at 22-weeks gestation presented with vaginal bleeding to an outside hospital. Her obstetric history was significant for 2 therapeutic abortions and 1 spontaneous abortion. During prenatal care, the fetus was noted to have a unilateral dysplastic kidney. The patient’s family history was significant for two family members with cervical cancer and two family members with endometrial cancer. She was placed on bedrest with inpatient admission. 48 h after admission she developed pelvic pain and uterine contractions. She expelled a mass vaginally measuring 11 × 9 × 5 cm with no fetal contents. The pathology from the outside facility showed a showed a highly cellular tumor composed of spindle-shaped cells and bizarre multinucleated giant cells with focal myxomatous change with mitotic count is greater than 50 per 10 high power field (Positive for CD10, SMA, ER, PR, EMA) with the differential diagnosis including endometrial stromal sarcoma and undifferentiated uterine sarcoma. The patient underwent examination under anesthesia, demonstrating a 3 × 3 cm defect of the posterior vaginal wall with active bleeding, which was sutured for hemostasis. The cervix was long, closed, and high with no evidence of bleeding and the fetus was intact with normal heart tones. She was discharged in stable condition.\nAt 27-weeks, the patient was referred to our institution for maternal-fetal medicine, and gynecology oncology consultation. On initial evaluation, she reported no prior abnormal gynecology history and no history of infertility issues. She denied dyspareunia and bulk symptoms prior to or during pregnancy, and her periods before pregnancy were normal. A pap smear at 27-weeks gestation was normal and negative for human papilloma virus. Her intake physical exam at our institution revealed a normal pelvic exam with no evidence of residual vaginal mass, and an ultrasound revealed no intrauterine myomas.\nGiven the differential diagnosis included an endometrial stromal sarcoma, an undifferentiated uterine sarcoma, and a primary vaginal sarcoma, the patient underwent magnetic resonance imaging (MRI) without contrast of the chest, abdomen, and pelvis at 27 weeks. MRI findings were notable for a peri-centimeter cyst in the cervix consistent with a Nabothian cyst, a multiloculated cystic structure in the fetal abdomen consistent with dysplastic kidney, and an anterior placenta. There was no evidence of metastatic disease.\nDue to the inability to ascertain whether the cancer had originated in the uterus, cervix or vagina, the patient desired definitive management for possible uterine sarcoma. In consultation with maternal fetal medicine and gynecology oncology specialists, the decision was made to proceed with a cesarean hysterectomy at 36 weeks. Betamethasone 12 mg intramuscularly was given for two doses 24 h apart with the first dose at 35 weeks and 6 days gestational age.\nAt 36 weeks and 1 day, she underwent planned exam under anesthesia of the vaginal canal with cesarean hysterectomy, bilateral salpingectomy, and peritoneal biopsies. She delivered a male infant weighing 2520 g with Apgars of 9 and 9. Operative findings were notable for normal appearing pelvic organs. There were no palpable or visibly enlarged pelvic/periaortic lymph nodes or evidence of metastatic disease throughout the peritoneal cavity. The neonate was admitted to the NICU post-delivery for prematurity. Neonatal US confirmed a right multi-cystic dysplastic kidney. He was transferred out of NICU on day 2 of life, and discharged home with his mother.\nThe final surgical pathology report was benign: no malignant or neoplastic cells were seen. The patient was seen at her 6-week postpartum visit and was doing well with a normal pelvic exam. At a follow-up surveillance visit 3 months postpartum, she was noted to have a 1 cm posterior vaginal wall lesion; biopsy results showed a high-grade sarcoma. The tumor was estrogen receptor and progesterone receptor positive. Computer tomography (CT) with contrast of the chest, abdomen, and pelvis showed no evidence of metastatic disease. The patient underwent an upper vaginectomy and proctoscopy. Operative findings were notable for 1.5 cm polypoid lesion in the posterior vagina wall 5 cm distal to the posterior vaginal apex. A 1 cm surgical margin was obtained circumferentially around this lesion where feasible, and surgical pathology showed high-grade sarcoma () of the polyp lesion with negative surgical margins. The tissue removed from this excision showed residual sarcoma, morphologically similar to the original pathology from the outside hospital pathology. All other vaginal biopsies were also negative for malignancy. Marker seeds were placed in the proximal and distal margins of the areas where the sarcoma was excised. She was treated adjuvant high dose rate vaginal brachytherapy using a multi-channel vaginal cylinder. She received a biologically equivalent 2 Gy dose (EQD2) of 45 Gy to the whole length of the vagina and 60 Gy to the post-operative bed (). A CT scan 36 months after surgery continue to show no evidence of disease. She remains disease free 58 months after completion of vaginal brachytherapy. |
A 65-year-old female patient developed chronic renal failure with unknown etiology and began hemodialysis in 2003. The patient denied past tobacco use or analgesic abuse. The patient received a renal transplantation in 2005 from a male cadaveric donor. Immunosuppressive therapy was applied using a triple immunosuppressive regimen, including cyclosporine (CsA), prednisone, and mycophenolate mofetil.The patient suffered from a pulmonary cytomegalovirus infection 8 months after the renal transplantation, and took oral cyclosporine and prednisone for a combination immunosuppressive treatment after the transplant. She had normal renal function, and the serum creatinine levels were maintained at 0.62 to 0.88 mg/dL. Forty-eight months after the renal transplantation, the patient went to the local hospital because of recurrent gross hematuria with waist pain on the left side. The CT examination revealed a left pelvic lesion of approximately 5 cm × 4 cm (Figure ). It was showed in the CT image that, irregular nodular soft tissue shadow filled in the left pelvis; the adjacent calyces and renal parenchyma were compressed; and there was no obvious swollen lymph gland shadow. Preoperative cystoscopy and chest computed tomography (CT) examinations revealed no abnormalities.A pathological examination showed that the size of the full-cut kidney and the surrounding fat capsule was 15 cm × 10 cm × 5 cm, and the actual kidney size was 12 cm × 7 cm × 4 cm. A bulging mass of 6 cm × 4 cm was observed at the renal pelvic cut surface without an envelope. This mass showed infiltrative growth that was gray and of fine quality, partly translucent and jelly-like, and also showed some hemorrhagic and necrotic areas. The renal parenchyma was atrophied and the ureteral length was 8 cm. Microscopic observations revealed two tumor components. One was a high-grade urothelial carcinoma component with partial papillary arrangement, and the epithelial cells were mostly heterotypic and pleomorphic, with some of them falling off. The cell nucleus was obviously pleomorphic, deeply stained, and the nucleolus was prominent and visible with pathological mitotic figures. The other was a heterologous sarcomatous component with a malignant peripheral nerve sheath tumor (MPNST) structure, and this was the dominant component. The tumors showed a diffuse growth or formed an alternate distribution of cell-rich areas and cell-sparse areas. The sparse areas were associated with myxoid stroma. The blood vessels were surrounded by tumor cells. The cells were fusiform and polygonal, and the cytoplasm was abundant, with red staining. The nucleus was large, deeply stained and irregular, and the nucleolus was prominent, with visible mitotic figures, multifocal necrosis and hemorrhage. Part of the region showed urothelial carcinoma and MPNST component with a transitional phase (Figure ). According to the TNM classification system, the patient was diagnosed with stage pT3N0M0.An immunohistochemical examination showed that cytokeratin of the urothelial carcinoma was positive (Figure ). Vimentin was positive in the sarcoma, and was negative in the urothelial carcinoma (Figure ). Some of the MPNST cells S-100 were positive in the carcinoma and sarcoma transition area, and strongly positive in the area of the sarcoma cells (Figure ). Smooth muscle actin (SMA) of sarcoma cells was negative, and Human Melanoma Black (HMB45) and desmin were negative.\nThe pathological diagnosis was that the left renal pelvic carcinosarcoma invaded the renal parenchyma, but was not involved the ureteral stump.\nThe patient accepted laparoscopic ureterectomy combined with lymph node dissection in our hospital. The operation went well, and 7 days after the operation, she was discharged from the hospital as she had recuperated. One week after the operation, she began to adjust the immunosuppressive regimen by using sirolimus (SRL) instead of cyclosporine, and the initial loading dose of SRL was 2 mg and 1 mg the next day, which was taken 4 hours after meals. Regular monitoring of blood concentrations was carried out after the conversion, and the drug dosage was adjusted according to the blood concentration, to maintain a plasma concentration of 4 to 6 μg/L of SRL. The patient refused to undergo any adjuvant therapy after the operation. She accepted regular postoperative follow-up. At the time of writing, 34 months after the left kidney ureterectomy, she is still alive with normal renal function. |
A 38 year old male married patient, medically free, referred to our center from a another hospital complaining from lower abdominal pain of few months duration, intermittent, vague, gripping in nature, associated with abdominal distention.\nHe didn't undergo any type of surgery previously. Past history of the patient was negative for any fever, sweating, vomiting, constipation, or weight changes. He also didn't suffer from obstructive urinary symptoms. Family history was negative too.\nOn examination, he actually was found to have large left, hypochondrial mass. It was painless & hard in consistency.\nOn investigation, his hemoglobin level and leukocytic count were within normal range. Markers for colon, liver and prostate cancers were negative. On computerized tomographic (CT) scan; there was a large heterogeneous mass measuring about 16 × 12 × 10 cm occupying the left hypochondrial region with multiple small metastatic right lung lesions (figures &). CT guided fine needle aspiration was performed and was positive for malignancy. At that time, patient was offered surgical intervention for the abdominal mass but he refused any further treatment. Upon follow up of the patient, CT scan was repeated 7 months later, there were multiple bilateral lung deposits the largest measuring about 1 cm in diameter. Also there was a large tumor in left atrium (figure ) extending to inferior left pulmonary vein measuring about 2 cm. the mass in the abdomen was still there as before. Then patient was sent home for tender care upon his request. Five months spent at home and the man came back again. He just reported a better feeling than before upon using some herbs (Teucrium polium, Cat Thyme) prescribed for him. On examination; there was no masses felt in the abdomen, so that CT scan was repeated. Unexpectedly; there was more than 60% reduction in the tumor size as well as disappearance of lungs & heart lesions as illustrated by figures &. During this interval, he was not given any type of radiation or chemicals as a therapy for his illness. Surgery was offered again and the mass removed by laparoscopic approach. Histopathologic analysis described a rounded hemorrhagic, necrotic and partially hyalized tumor that is markedly vascular and has a very prominent alveolar pattern. Immunohistochemical staining including cytokeratin, desmin, actin, S-100, synaptophysin, HMB45, chromogranin, vimentin, myoglobin and PAS was performed. All stains were negative apart from PAS and myoglobin which were positive (figure ). The overall picture was consistent with alveolar soft part sarcoma (ASPS) infiltrating the omentum. Similarly, endoscopic assessment of upper gastrointestinal tract (due to epigastric pain) demonstrated multiple sessile polyps in stomach & duodenum; biopsies were taken, and the diagnosis was alveolar soft part sarcoma. Few months' later, patient developed headaches, brain CT scan was done and unfortunately he was found to have metastatic brain lesions. He was referred to a specialized center for radiotherapy and died within 6 months. |
A two-day-old female infant, born at term after uneventful pregnancy, was referred to our paediatric cardiology team to be evaluated for a light murmur. She was born by vaginal delivery; her post-natal adaptation was normal and auxometric parameters were adequate for her gestational age. No clinical signs of dysmorphism or cyanosis or dyspnea were evident; her peripheral oxygen saturation was 98% at room air and the heart rate was normal. On cardiac auscultation, a 1-2/VI murmur could be heard in the left upper sternal border; pulmonary auscultation as well as peripheral pulses were normal. Colour-Doppler echocardiography revealed the absence of the right pulmonary artery, whereas the main and left pulmonary arteries had normal size. The pulmonary valve was normal. There was no evidence of a patent ductus arteriosus (PDA) nor other cardiac defects except for a small patent foramen ovale. The size and function of both cardiac ventricles were normal and without ventricular hypertrophy. The size and origin of the coronary arteries, as well as the aortic arch, were normal. A physiological tricuspid insufficiency was noticed, with an estimated pulmonary artery pressure of 30 mmHg. These cardiovascular features were confirmed by echocardiographic examination, 1 week later. Suspecting an isolated unilateral absence of the right pulmonary artery (UARPA), we decided to carry out chest X-ray and MRI. The former showed normal pulmonary aeration with regular size of both lungs and a normal cardiac silhouette, the latter showed the absence of the right pulmonary artery, with a slightly increased lumen and size of the main and left pulmonary arteries (Fig. ). Fortunately, the right lung was shown to be perfused by some systemic collateral arteries arising principally from the right subclavian artery and from the celiac trunk. MRI also excluded other heart defects and confirmed normal cardiac function.\nIn the light of an isolated malformation, as our child was still asymptomatic and she had regular growth rates, no medical or surgical treatment was deemed to be needed. Nevertheless, as symptoms may occur later in life, a thorough clinical and cardiological follow up was immediately started. At present, our patient is 3 years old and remains completely asymptomatic. She is a very active child and shows no sign of fatigue. She has not had any feeding difficulties and her growth has been regular. No major pulmonary infections have been reported. During her first 3 years of life, we have thoroughly monitored her pulmonary artery pressure and the right ventricular function, which have always been normal. In the light of these findings and her clinical condition, our referral paediatric heart surgeon also recommended a wait and see attitude. |
The proband was a 6-year-old girl born to non-consanguineous parents. The pregnancy and the delivery were normal. She began to speak and walk independently at the age of 19 months and had a normal head circumference. The seizures occurred at the age of 8 months and 1 year, respectively, presenting with eyes rolling involuntarily and lips cyanosis, but there were no seizures in the next 2 years. Until the age of 4 years, her symptoms evolved into dystonia, majorly with the head backward, stiffness of the right lower limbs, and hallux dorsal flexion. Occasionally, her right arm was rotated inward and could not be flexed, accompanied by crying and shaking of the whole body. At the early stage, these symptoms were mild, with a frequency of three to eight times per month, each lasting about 1 to 2 min. The electroencephalogram (EEG) showed sharp waves and sharp slow waves in the right central temporal and left central region with a normal background during wake and sleep. Oral oxcarbazepine was then administered, but the attacks were not controlled completely. Since the age of 5 years, the frequency and duration of the attacks increased, ranging from once every 2 to 3 days to twice to thrice a day and lasting up to half an hour each time. Additionally, she experienced frequent paroxysmal hypokinesia with low muscle tone, which were relieved after resting. Nervous system physical examination was performed during the interictal period, and there was no abnormal muscle tension or strength and no positive ataxia signs, while the Assessment of the Child Development Scale indicated that her development was lagging behind mildly. The metabolic screening and other blood examinations were normal. KD was introduced along with oral oxcarbazepine when she was 5 years old, and it was tolerated well during the follow-up. Up to now, she has been free of these symptoms for nearly a year. The EEG returned to normal after initiating KD for 2 months. |
A 71-year-old male patient with a persistent central corneal epithelial defect and stromal thinning of 5 mm diameter on his right eye with no evidence of infection or corneal neuropathy was intermittently responsive to conservative topical treatment with antibiotic drops and ointment, intensive lubricating drops, and bandage contact lenses. The central cornea had a thinner area of 2 mm diameter with only 1/3 of the stroma remaining before Descemet's membrane and showed no sign of healing with only intermittent epithelialization despite the conservative treatment for over eight weeks from presentation. There was no evidence of corneal hypesthesia, and repeated corneal swabs for microbiology investigation proved the sterility of the lesion. The clinical suspicion of corneal rubbing against the pillow because of his sleeping preference on his right side was the most probable cause of his condition.\nAs counselling for his sleep pattern and conservative lubricating treatment had no effect, we proceeded with surgical management of his corneal defect using cryopreserved AM. We followed our previously described standard technique for corneal surface reconstruction with the membrane placed over the whole cornea [].\nFollowing peribulbar and subconjunctival anaesthesia, we carefully debrided the thinner crater and scraped off the corneal epithelium up to the limbal margin followed by a 360° conjunctival peritomy (Figures and ). Small cut pieces of AM were used to fill the stroma defect level with the surface, and a larger piece was trimmed to cover the whole corneal and limbal area with the epithelial side down used (“sticky” stroma side up) as a patch for a healthy corneal epithelium to grow underneath. We secured the membrane under the conjunctival edges with a continuous 10.0 nylon suture (). Finally, we applied a subconjunctival injection of a mixture of dexamethasone and gentamycin and placed a bandage contact lens for 2 to 3 weeks (). Our routine postoperative regimen was combined steroid and antibiotic eye drops 6 times daily for two weeks tapering down thereafter.\nFor deep corneal ulceration, we use small pieces of AM to fill the stroma defect placed as grafts with epithelial side up aiming at subepithelial or transepithelial integration of the membrane in relation to the newly formed epithelium. Following limbal peritomy, a large piece of AM is placed as a patch with the epithelial side down covering the entire cornea and limbal area, aiming at superficial localization (disintegration) of this covering piece of amniotic membrane. In theory, this arrangement will facilitate the growth of a new, healthy corneal epithelium from the intact limbal area under the superficial layer of AM but over the small pieces of AM covering the stroma defect [, ]. We have abandoned the technique of using a smaller than the corneal diameter piece of AM as a graft (epithelium up) which is sutured on to the cornea within the limbus so that the newly formed epithelium will grow over the membrane as less effective and more traumatic for the cornea [].\nThe AM graft was prepared and cryopreserved by our local eye bank using the Good Tissue Banking Practice procedures []. Despite the tested sterility of the ocular surface, the preoperative antibiotic treatment, and the uneventful perioperative period, he developed a 2 mm hypopyon within 48 hours when we reviewed him after primary AMT (). He was using our routine regimen of tobramycin 3 mg/ml and dexamethasone 1 mg/ml eye drops six times daily on a fixed combination.\nDespite the alarming clinical presentation, there were no anterior chamber reaction and no signs of active infection; therefore, we considered the hypopyon formation being a sterile toxic or immunologic reaction. To confirm this diagnosis, we took a surface swab from the membrane and performed a full microbiological examination of the residual membrane and its culture medium which we always keep for up to 7 days for quality control which were proven negative.\nThe patient continued with our standard post-AMT treatment with increased dosage every two hours for three days, followed by 6 times daily for two more weeks tapering down thereafter. The hypopyon resolved completely with the current treatment a week later with no further complications or anterior chamber reaction ().\nThe epithelial defect and corneal thinning healed satisfactorily with smooth epithelium and a visible stromal scar with no recurrence or hypopyon 5 weeks postoperatively (Figures and ). |
A 55 year old man was diagnosed with anemia by his family physician towards the end of 2017. In February of 2018, an upper GI endoscopy and colonoscopy were performed which showed a right colon cancer. CEA was 40.4. CT of the chest, abdomen, and pelvis was obtained in February of 2018. This showed a mass 7 cm in greatest diameter that was immediately adjacent to the undersurface of the right liver and contiguous with numerous loops small bowel and the lowest part of the second portion of the duodenum (). There were mildly prominent mesenteric lymph nodes but no evidence of distant metastatic disease.\nIn February of 2018, the patient underwent a single port laparoscopic right colon resection. The surgeon described the tumor as stuck to the undersurface of the liver with adhesions taken down without difficulty. On histopathologic examination, the tumor was PT3N0M0 with 0/16 positive nodes. Perineural invasion was identified. The cancer was moderately to poorly differentiated. Systemic chemotherapy was not recommended.\nIn June of 2018, the patient began noting problems with digestion and pain after eating. CEA had increased to 940 ng/ml. Repeat CT scan showed multiple nodules within the greater omentum compatible with peritoneal metastases. The liver, kidneys, and ureters were normal. There was marked stranding within the right colon resection site (). Bowel loops proximal to the prior ileocolic anastomotic site were moderately dilated and fluid-filled. A mass was noted in the abdominal wall at the laparoscopic port site. There was also a mass in the rectovesical space immediately adjacent or invading the right and left seminal vesicles.\nIn July of 2018, the patient underwent exploratory laparotomy. The posterior surface of the liver was layered by cancer. The undersurface of the right hemidiaphragm was covered by tumor nodules. The pelvis contained a 4 cm tumor mass invading into the seminal vesicles. The old ileocolic anastomosis was involved and needed to be resected. The umbilical port site was infiltrated by cancer. Near complete resection was possible by cytoreductive surgery.\nIntraoperatively, the patient was treated with HIPEC-melphalan []. This postoperative course was unremarkable. The histopathology showed adenocarcinoma compatible with the primary tumor in all tissues submitted. The peritoneal cancer index was 28 and the CC score was 2 []. The resection was judged to be a palliative effort and its major goal was to relieve intestinal obstruction and to prepare the patient for rapid initiation of palliative systemic chemotherapy.\nNeither of our patients was evaluated by the MDT preoperatively. Their preoperative and intraoperative management was evaluated in an attempt to improve the outcome of subsequent patients. |
We report a case of a 37-year-old professional male athlete presenting with a seven-month history of worsening respiratory function. He was diagnosed with asthma and managed in the community. He was referred for further investigation following worsening of his symptoms and the onset of stridor. Flexible nasendoscopy revealed intact vocal cord function with a mass lesion visible in the trachea. Rigid bronchoscopy reported an intraluminal tracheal mass immediately inferior to the cricoid extending five centimeters caudally resulting in eighty percent tracheal obstruction (). The carina and upper oesophagus were noted to be grossly free of disease. A biopsy of the mass diagnosed tracheal adenoid cystic carcinoma of cribriform and tubular variant.\nMRI scan and PET-CT demonstrated a low to intermediate FDG uptake of 5.9. Key findings from imaging included submucosal extension within the tracheal lumen, invasion of thyroid gland, and no direct invasion of the cricoid cartilage. There was no evidence of cervical nodal enhancement or distant dissemination. The patient's case was discussed at our multidisciplinary team meeting with input from cardiothoracic surgery, medical oncology, radiation oncology, and the head and neck team. Tracheal resection with primary cricotracheal anastomosis, total thyroidectomy, and preservation of the larynx was the proposed surgical intervention.\nT-shaped neck incision at the level of cricoid cartilage was made with a partial sternotomy to afford greater access (). Vessel loops were applied to the innominate artery and vein. Mobilization of the thyroid lobes was then performed with identification and preservation of the recurrent laryngeal nerves bilaterally. A small pocket of disease was identified bilaterally at the cricothyroid joints surrounding both recurrent laryngeal nerves. Inspection was then made of the distal aspect of the dissection where submucosal extension of disease in the trachea was also identified tracking caudad towards the carina. A curative resection was clearly not a possibility. A 5 cm resection of the trachea was performed to achieve gross intratracheal disease clearance. Following the tracheal resection a cricotracheal anastomosis was performed, using 3-0 polydioxanone sutures preserving the recurrent nerves at the cricothyroid junction. The tension on the cricotracheal anastomosis was reduced by suprahyoidal muscle release and its strength augmented using Tisseal on the anastomotic line. Postoperative care included suturing of the patients chin to his chest with 0-nylon sutures, ensuring the patient's head was kept flexed and thus reducing tension on the new anastomosis. An alternative to this method is with the use of a custom made neck brace. The patient was sedated and intubated in intensive care unit for 7 days prior to extubation in operating theatre. Mild surgical emphysema was noted but resolved by day 3 after operation. The patient was also treated with broad-spectrum antibiotics, a tapering dose of corticosteroids and continuous humidified oxygen and saline nebulisers.\nChin to chest sutures were removed at day 14 after operation and patient was discharged home. Three weeks post-op the patient maintained normal voice quality, normal deglutition and showed significant improvement in exercise tolerance.\nHistopathological analysis of the operative specimen confirmed adenoid cystic carcinoma with a predominately cribriform pattern (). The disease involved multiple tracheal rings with extensive perineural and lymphovascular invasion. The tumor invaded the thyroid gland and both the superior and inferior margins were positive for tumor involvement.\nFollowing discussion at our MDT meeting a decision was made to give adjuvant radiation and concurrent platinum based chemotherapy given the potential locoregional advantage its inclusion may afford []. A total radiation dose of 60 Gy and a cisplatin dose of 30 mg/m2 were administered under the care of the medical and radiation oncology team. No severe or serious adverse events were reported.\nA follow-up PET scan performed 3 months after adjuvant radiation and chemotherapy showed no FDG uptake both locoregionally and distally. The patient has also returned to playing football at professional level maintaining an excellent level of pulmonary function, phonation, and deglutition. |
A 52-year-old male was admitted to an outside institution with lethargy, weight loss of 30 kg, and severe unexplained anemia (hemoglobin 4.5 g/dL; hematocrit 15%). The patient felt better after transfusion of 4 units of packed red blood cells, and gastroscopy performed at the time revealed a healed duodenal ulcer and erosive gastritis.\nWhen he lost his medical insurance, he was discharged and advised to have further workup performed elsewhere. During a difficult colonoscopy at a charity clinic, a large tumor was found in his right colon that precluded passage of the scope to the cecum (see ). Biopsy confirmed an infiltrating adenocarcinoma. Polyps in the sigmoid and transverse colon were also removed and found to be tubular adenomata. The patient was then referred to our institution, a safety-net hospital, where a CT scan was performed. The imaging demonstrated a 5 × 5 cm mass in the mesentery with spiculated calcifications, as well as an additional mass near the ileocecal valve that had the appearance of an intussusception (see ). Malrotation was not suspected, although later review of the imaging with a specialized CT radiologist demonstrated inversion of the normal SMA to SMV configuration (see ). Malrotation was not suspected. The liver was free of metastases. His past medical history included no prior surgery.\nA laparoscopic right hemicolectomy was scheduled and commenced with introduction of a 10 mm optical trocar and two 5 mm ports. Upon entry, most of the small bowel was found plastered to the right flank, and the right colon was not visible. The left colon was visualized, but even after releasing adhesive bands holding the small bowel to the right side and mobilizing these loops to the left of midline, the ascending colon was not visible. It was decided that laparotomy was required to elucidate the findings. The abdomen was entered through a midline incision, and after mobilizing and packing off the small bowel to the left, the right paracolic gutter was found to be empty. The duodenojejunal junction, including the entirety of the duodenum, lays to the right of the vertebral column. The right colon was tethered to the side wall of the abdomen on the right by long bands (Ladd's bands), which were eventually divided to obtain adequate mobilization. The cecum and appendix were found to occupy the left upper quadrant (see Figures and ). A tumor was palpated in the ascending colon, and a firm mass in the mesentery to its left was thought to be metastatic nodes. The superior mesenteric vessels appeared to have their usual orientation. The right colon was thereby mobilized beyond the left branch of the middle colic, and a right hemicolectomy was completed. The patient made an uneventful recovery.\nPathology revealed a 5 cm infiltrating adenocarcinoma of the ascending colon and metastatic tumor in the mesentery with no evidence of lymphatic tissue involvement. 23 lymph nodes removed were negative for metastases. The patient received chemotherapy and is doing well 12 months after surgery. |
A 15 years old girl presented to the outpatients' clinic accompanied with her aunt; her primary complaint was that her left arm was hurting and she was unable to move it. These symptoms started about 4 weeks ago and gradually increased in severity until there was no function left in the affected arm. She denied any conflict or stressor prior to the onset of symptoms; further psychiatric evaluation revealed that she had a low mood with loss of pleasure in day to day activities. She had a decreased appetite with initial and middle insomnia, diminished energy and easy fatigability. She expressed ideas of reduced self worth and helplessness but denied having suicidal ideation. She also had severe irritability and increased interpersonal sensitivity. Physical examination including a detailed neurological examinational was completely normal.\nShe was diagnosed as suffering from conversion disorder with comorbid major depressive disorder with mixed features according to the Diagnostic and Statistical Manual of Mental Disorders, fifth edition (DSM-5; American Psychiatric Association, May 2013). Psychopharmacological treatment was started and consisted of escitalopram 10 mg/d, mirtazapine 30 mg/d, and levetiracetam 1,500 mg/d. She was seen regularly on a weekly basis and supportive psychotherapy and physiotherapy sessions were also included. The first 6 weeks resulted in modest progress with improvement in sleep, some relief of depression, decreased pain in the affected arm with increased range of motion. However, in the next 4 weeks the patient had anxiety and restlessness with mood lability and her progress stalled. At this stage aripiprazole was introduced in a dose of 10 mg/d and in the following 3 to 4 weeks there was a remarkable betterment in her condition with remission of the depressed mood and almost complete return of function of the left arm with only minor residual pain on full motion. The period of follow-up was increased to once every 3 weeks and the following medications were continued to maintain the remission phase of the mood disorder: 1) Aripiprazole 10 mg/d; 2) Escitalopram 10 mg/d; 3) Mirtazapine 30 mg/d; 4) Levetiracetam 1,500 mg/d. |
Mr P was a married man in his 40s with one child. He had served in the UK army and was deployed to the Bosnian conflict in the mid-1990s. During his deployment he was stationed at a morgue that had been set up to aid the identification of bodies recovered from a mass burial site. He described how the morgue contained a large number of bodies in various states of decomposition, with varying degrees of physical trauma. He reported re-experiencing fragmented intrusive images of the morgue and daily emotionally distressing nightmares of which he could not recall the content. He experienced high levels of anxiety that triggered his dissociative symptoms. These appeared to function as a coping strategy to avoid distressing emotions connected to his memory of the morgue. Other avoidance symptoms included feeling detached from people around him and feeling emotionally numb. Mr P avoided family occasions and busy places which could trigger his hyper-arousal symptoms and had never spoken to anyone about his experiences. He reported coping historically through the use of alcohol to block out his emotions. His avoidance behaviours had maintained his PTSD symptoms.\nAt the start of therapy sessions, Mr P was reluctant to talk about his past experiences stating he only felt able to discuss vague details about his intrusive memories. Mr P attempted to use physical objects such as stones and leaves, or his e-cigarette, as grounding strategies in the clinic room. However, he also reported feeling unable to tolerate any emotions connected to the events and found it difficult to identify peri-traumatic cognitions. Mr P appeared to experience intrusions of one particular fragmented image and could not recall any other contextual information from before or after this moment.\nIn both cases Mr C and Mr P experienced difficulties in engaging with traditional methods of imaginal reliving and prolonged exposure, due to high levels of avoidance and low thresholds for dissociation, and subsequently had difficulties accessing their cognitions for updating. For both clients the prior use of multiple grounding methods in the therapy room were unsuccessful. They experienced their traumatic memories as fragmented images with a sense of ‘nowness’ (), which indicated a distinct lack of contextualization.\nAn adaptive approach was then trialled to address these hindering factors. This involved three stages: (1) walking the client through the imaginal scene outdoors to address dissociation, (2) viewing the imaginal scene from multiple perspectives to facilitate contextualization of the memory and (3) identifying and reappraising the cognitions with frequently used approaches in trauma-focused therapy. |
A 45-year-old male patient was reported to the Department of Maxillofacial Prosthodontics, with a chief complaint of missing right eye and wants it to be replaced with an artificial prosthesis []. Patient had given a history of trauma to the right eyeball due to an accident 1 year back. Patient had given a history of surgical enucleation of the right eye under general anesthesia 15 days after ocular trauma. When further asked, we come to know that the enucleation was only done and no intraorbital implant was placed and also there was no history of using ocular prosthesis. Examination of the socket revealed an adequately healed defect with the absence of eyeball, the presence of superior sulcus deepening, narrow opening of eye with upper eyelid ptosis, depth of the lower fornix was reduced, and movements of the musculature were normal. A thorough inspection and palpation of the defect were done to check the resiliency of the superior and inferior palpebrae. On clinical examination, there was the absence of swelling, bleeding, inflammation, and pain in relation to the anophthalmic socket. On thorough examination of the socket, it was found that there was no undercut present.\nThe treatment plan was involved, fabrication of an ocular prosthesis with the modifications to correct the opening of both eyelids with correction of ptosis, expansion of remaining eye socket and the superior sulcus deformities. It was planned that a custom-made ocular prosthesis would be given to meet the needs of the patient. The treatment planned and technique involved was explained in detail to the patients along with the limitation of the technique and a written consent was obtained from the patient.\nThe contralateral eye measurements were taken as a guide for the fabrication of identical custom-made eye prosthesis. The measurements from the inner canthus to the outer canthus was 3.2 cm, inner canthus to the center of pupil was 1.9 cm, center of the pupil to the outer canthus was 1.6 cm, bridge of the nose to the inner canthus was 1.4 cm, diameter of the cornea was 1.1 cm, length from the center of pupil to the lower eyelid was 0.9 cm, and center of the pupil to the outer limbus was 0.6 cm.\nPrimary impression of the defect was made with alginate (Zelgan, Dentsply, Gurgaon, India). Beading and boxing of primary impression was done, and cast poured in type II dental plaster (Kalabhai, Mumbai, India). Wax spacer with a syringe was positioned in the center of the defect on the cast, and a special tray was fabricated using cold cure clear acrylic resin (DPI, Mumbai, India) []. The syringe fitted to the special tray was filled with polyvinylsiloxane monophase impression material (Reprosil, Dentsply, Milford, USA), and the tray was placed over the defect, and the syringe was pressed to inject the impression material into the orbital socket []. Instruction was given to the patient to do the movements of the eye to record the functional impression of the orbital socket. The impression was removed and checked for any bubbles or inaccuracy. The beading and boxing of the impression was done in such a way that a split cast mold in two sections was obtained []. Wax pattern was fabricated in the defect area of the sectioned and stabilized cast []. The color matching of the stock eye shell's iris was done with the contralateral side of the iris. Iris part is trimmed from the stock eye shell and was positioned in scleral wax pattern with the help of measurements taken from contralateral eye []. Try-in of the wax pattern was done to evaluate the size, comfort, the simulation of eye movement, and the eyelid support []. Once the patient was satisfied, the iris was removed from the wax pattern. Flasking and dewaxing of the wax pattern were carried out, and packing was done using tooth-colored heat cure polymethyl methacrylate (PMMA) resin (Pyrex, Roorkee, India). The iris was placed back in the position using adhesive (Fevikwik, Pidilite, Mumbai, India) in the scleral shell, and characterization of the eye was completed using floccules and artist's oil paints (Camlin, Mumbai, India).\nThe characterized eye was coated with a thin layer of inlay wax (Shiva, Thane, India) and again flasking and dewaxing were done and was packed with heat cure clear acrylic resin so that all the floccules and oil paints are retained on the sclera, and this clear acrylic simulates the conjunctiva and curing was done. The ocular prosthesis obtained was trimmed and finished []. The insertion of the eye prosthesis was done, and postinsertion instructions were given to the patient []. Prosthesis should be removed from the eye socket during night times and while sleeping. Prosthesis should be cleaned before placing and after removal from the socket. Lubricate the eye with Lubrex eye drops once daily. Clean the prosthesis with mild soap solution once in a week and recall visit for every 6 months. |
A 2-month-old baby girl weighing 3.5 kg presented with a huge swelling on the left side of her neck. The swelling was small in size when noticed at birth, which progressed gradually to the present size [Figures and ]. The child presented with a huge swelling with restricted mouth opening and difficulty in feeding. Examination revealed swelling on the left side of the neck sized 10 cm × 10 cm, which was cystic, non-tender and extending from the angle of the mandible to the clavicle. The skin over the swelling looked normal with no local rise of temperature. The swelling was huge and presented with difficulty in swallowing. In anticipation of rupture of the swelling and aspiration of its contents and also because it could lead to airway obstruction, it was decided to operate the child.\nPre-operative evaluation was thoroughly carried out. Evaluation for concurrent anomalies like Down's syndrome, Turner syndrome or congenital heart defects was done. The size and extent of the neck mass was defined carefully in an effort to detect the potential for airway compromise and to avoid soft tissue trauma during intubation.[] Chest X-ray was done to exclude the presence of chest infection and intrathoracic extension of the tumour.\nA rescue tracheostomy by the surgeon was available as a standby during induction.[]\nBecause cystic hygroma presents with difficult airway challenge to the anaesthesiologist, a difficult airway cart was kept ready. The child was premedicated with intravenous (IV) atropine 70 mcg. A shoulder roll was used to keep the child at optimal laryngoscopic position as the child had a larger occiput compared with the rest of the body. Because a larger tongue in the child could obstruct the airway after induction, an inhalational induction was considered.[]\nAs sevoflurane was not available in our institute, the child was induced with halothane in oxygen by face mask. A straight blade laryngoscope was used as children have an anterior, cephalad placed larynx and short and stout epiglottis.[] Halothane does not have a noxious smell and is still commonly used for the gaseous induction of anaesthesia in places where budgetary concerns limit the use of sevoflurane.[] At first attempt, laryngoscopy was difficult as the vocal cords were not visualised and only the epiglottis could be visualized, which was shifted to the right side due to displacement of the soft tissues. The child was ventilated with mask and a second attempt of laryngoscopy was made. This time the glottis could be seen after shifting the soft tissues towards the left side by the assistant and the trachea was successfully intubated with uncuffed endotracheal tube (ETT) size 3.5 mm, and it was fixed just 1 cm beyond the vocal cords in order to avoid accidental extubation. Anaesthesia was maintained with 66% nitrous oxide in oxygen with controlled ventilation with Jackson Rees modification of Ayre's T piece. Inj. Fentanyl 8 mcg and Inj. atracurium 2 mg was administered and supplemental doses 0.1 mg/kg were used as and when necessary. Blood loss throughout the surgery was calculated to be about 100 ml and was replaced. The cysts were multiloculated and excised completely. All the vital signs were stable in the intra-operative period. At the end of surgery, the child was reversed with Inj. Neostigmine 0.05 kg/kg and Atropine 0.01 mg/kg. In view of possible collapse of trachea and obstruction to airflow, it was decided not to extubate the trachea in view of the difficulties faced during intubation.\nThe child was shifted to the Neonatal intensive care unit and was extubated on the second post-operative day after return of adequate muscle power, respiratory efforts, cry and movements. |
A 50-year-old female, with a history of hypothyroidism and constipation and with a family history of a mother with multiple colonic polyps, presented for a routine screening colonoscopy. During the colonoscopy, dozens of polyps were identified throughout the colon. Many were removed, including 8 polyps between 3 and 10 mm from the cecum, 12 polyps between 3 and 10 mm from the ascending colon and one 6-mm polyp from the transverse colon (). Many residual polyps were left behind due to high burden of disease. Internal hemorrhoids and diverticulosis of the sigmoid colon were also noted. Of the 21 polyps removed, 20 were tubular adenomas and 1 was a lymphoid polyp. Given the unusual findings, the patient returned to clinic a few days later.\nIn clinic, a thorough history was performed. The patient’s only gastrointestinal history was chronic constipation, with bowel movements every day to every other day, sometimes requiring laxatives. She otherwise denied rectal bleeding, diarrhea or weight loss. She confirmed no family history of colon cancer, but her mother reportedly had many colon polyps. Her father passed away at an early age in a motor vehicle accident. None of her three healthy siblings or child had gastrointestinal issues. Given the high number of polyps in both the patient and mother, without significant personal or family history, she was referred for genetic testing.\nGenetic testing returned positive for an AXIN2 mutation, which has been associated with oligodontia-colorectal syndrome. She again returned to clinic, where further investigation revealed that she lacked eight permanent teeth, requiring placement of a bridge and implants as a child. She recalled her mother having dental issues as well. She did not believe anyone else in her family had dental anomalies.\nIn addition to genetic counseling, she was recommended to undergo a follow-up colonoscopy. On repeat colonoscopy, 31 sessile polyps were found throughout the colon and were removed, still with multiple polyps left behind ().\nGiven the presence of an Axin2 mutation, her dental history and colonoscopy findings and her presentation not matching any known colorectal cancer syndrome, it was felt that she had or was at risk for oligodontia-colorectal cancer syndrome. However, there are no current diagnostic or management criteria. It was felt that, without intervention, she would likely need frequent surveillance colonoscopies and polypectomies to remove potentially precancerous polyps. Given this prospect, she was referred to a general surgeon and was ultimately elected for a subtotal colectomy with ileorectal anastomosis, as sparing the rectum would result in less bowel dysfunction. The rectum also did not have any polyps on previous colonoscopies and could easily be surveyed with sigmoidoscopies.\nFollowing surgery, the patient had an unremarkable post-operative course and was discharged on post-operative day 3. One year following surgery, the patient had improved bowel function, with two to three bowel movements per day without the use of laxatives. A flexible sigmoidoscopy was negative for polyps in the rectum, with the plan to repeat a flexible sigmoidoscopy in 1 year and then to subsequently space out surveillance. To date, the family has not yet undergone genetic testing. |
The patient discussed herein is a 60-year-old male who presented to the surgical oncology clinic with a one-year history of intermittent abdominal pain. He was previously evaluated by gastroenterology at an outside institution and had undergone a CT scan and an endoscopic ultrasound (EUS). This imaging revealed an approximately 4.5 cm mass between the duodenal bulb and the head of the pancreas with both solid and cystic components. A biopsy was performed during the EUS, but a tissue diagnosis could not be made.\nAt the time of our initial interview, the patient reported persistence of his abdominal pain as well as 15 pounds of weight loss over the preceding three months. He was otherwise asymptomatic. He admitted to occasional smoking and alcohol use. His family history was negative for GI malignancy. On exam, he was noted to be anicteric and nonjaundiced. Abdominal exam was normal aside from a reducible nontender 3 cm umbilical hernia.\nAfter the patient's office visit, an MRI was performed for better characterization of the previously identified mass. This showed a 4.4 cm predominantly solid mass in the bulb and second portion of the duodenum with extension into the pancreatoduodenal groove as well as fluid in the right pararenal and paraduodenal spaces (Figures and ). A repeat endoscopy with EUS was performed. Endoscopy showed a nodular, edematous, friable mass in the first portion of the duodenum causing mild stricture of the lumen (). EUS revealed a slightly nodular, ill-defined, hypoechoic and heterogeneous submucosal mass in the medial wall of the first portion of the duodenum extending into the duodenal sweep and causing mild stricture. The duodenal wall was thickened with loss of demarcation between layers (). Repeat biopsy and fine needle aspiration (FNA) of the mass taken at the time of EUS revealed only duodenal mucosa with dilated lymphatic channels and prominent lymphoid follicles.\nGiven the remaining diagnostic uncertainty, the patient was discussed in a multidisciplinary fashion at our GI tumor board. It was ultimately decided that the patient should undergo surgical resection for definitive diagnosis. A pancreaticoduodenectomy was performed without intraoperative complication. The patient's postoperative hospitalization was prolonged due to the ileus which was managed with NGT decompression. This appeared to resolve by postoperative day 8 at which point he was discharged. The patient was readmitted two weeks later for severe hiccups and vomiting. He was treated for gastroparesis with erythromycin and Reglan and discharged on hospital day 4. On postoperative follow-up at one and six weeks, the patient reported tolerance of regular diet and freedom from hiccups and nausea.\nGross and microscopic pathologic findings of the surgical sample were suggestive of PP (Figures and ). The mass effect in this case was secondary to widespread fibroblastic proliferation and local edema (). Immunohistochemical stains were negative for beta-catenin, anaplastic lymphoma kinase- (ALK-) 1, and CD-117, which ruled out fibromatosis, inflammatory myofibroblastic tumor, and gastrointestinal stromal tumor, respectively. There was no elevation of serum IgG4 or significant amounts of IgG4-positive plasma cells in the tissue, ruling out autoimmune pancreatitis and IgG4 disease. |
The patient is a 15-year-old Caucasian female who was diagnosed with CRMO in 2007 at 5 years in the context of right thigh pain. provides a timeline of the patient's symptoms and course of disease. Magnetic resonance imaging (MRI) showed multifocal abnormal bone marrow signal in the right femur, the left femoral neck, and the proximal epiphysis and metaphysis of the right tibia, which was associated with osteolysis and callus formation (). Radio-nucleotide bone scan found increased uptake involving the left sacrum, left proximal femur, and femoral neck region as well as the midshaft of the right femur and the proximal right tibia. Bone biopsy of the lytic lesion involving the midshaft of the right femur was not consistent with malignancy and showed red blood cells and scattered neutrophils and lymphocytes. Over the next several years (2007–2015), she was followed by orthopedic surgery and was treated only with intermittent ibuprofen as needed for pain. She was noted to have a leg length discrepancy at the age of 7 years, and in 2014, at the age of 12 years, she had surgery to fuse the growth plate to prevent right leg growth (right leg was 4.5 cm longer than her left leg at that time).\nShe established Rheumatologic care in March 2015 at age 13, and right quadriceps muscle atrophy and failure to thrive with a weight and height under the third percentile were noted at this time. Bone scan revealed increased uptake in the right femur and asymmetry of activity in the growth plates of the knees and ankles with decreased activity in the right side compared to left. She was treated with naproxen 250 mg twice daily (8.7 mg/kg BID) and prednisone 20 mg per day (0.7 mg/kg), tapered by 5 mg weekly. Two months later, prednisone was discontinued, and she continued to have good control of her leg pain on NSAID monotherapy. After a few months, she developed new diarrhea with vomiting and weight loss. Her blood pressure was elevated, and a renal ultrasound and electrocardiogram were unremarkable.\nShe was referred for Gastroenterology evaluation and was found to have positive stool occult blood along with a perirectal skin tag. Laboratory studies revealed anemia with a hemoglobin of 7.1 g/dL (nl 12–16 g/dL), thrombocytosis with platelets of 744 k (nl 150–450 k), erythrocyte sedimentation rate (ESR) of 69 mm/hr (nl 0–10 mm/hr), and C-reactive protein (CRP) of 129 mg/L (nl 0–3 mg/L). There was no family history of autoimmune disease, and tuberculin testing was negative. Colonoscopy revealed pancolitis with crypt inflammation and crypt abscesses with no granuloma consistent with ulcerative colitis (). NSAID therapy was discontinued, and treatment for UC with prednisone 1 mg/kg/day (40 mg) tapering by 5 mg weekly along with sulfasalazine was initiated. Infliximab 3 mg/kg infusions were added the following month.\nAt the visit for her second infliximab infusion, she presented with tachycardia, a blood pressure of 230/190, and headache and was admitted to the pediatric intensive care unit for hypertensive emergency requiring nicardipine infusion. Her examination was notable for right-sided Horner's syndrome, decreased right leg pulse pressure, and an abdominal bruit. Echocardiogram showed a small pericardial effusion with reduced left ventricular ejection fraction. Laboratory studies revealed negative ANA and ANCA screens, normal C3 and C4, and normal von Willebrand factor antigen. CT angiogram (CTA) of the abdomen and pelvis showed narrowing of the mid-aorta, proximal renal arteries, celiac artery, and superior mesenteric artery (Figures –). CTA of the chest showed marked descending thoracic and abdominal aortic wall thickening with progressive luminal narrowing and wall thickening of the visualized portion of the right common carotid artery and celiac trunk, enlarged left atrium, left ventricular hypertrophy, and a small pericardial effusion (). MRI/MRA brain showed anterior and posterior circulations of the brain were without occlusion or aneurysm with the patent carotid and vertebral arteries of the neck (Figures and ). Given these findings, she was diagnosed with TA complicated by middle aortic syndrome. She was started on metoprolol 50 mg daily (1.5 mg/kg daily), amlodipine 5 mg daily, famotidine 20 mg twice daily, aspirin 81 mg daily, and increased dose and frequency of infliximab from 3 mg/kg every 8 weeks to 5 mg/kg IV every 4 weeks along with prednisone 20 mg daily (0.6 mg/kg daily) and mesalamine 1 g BID. The patient continued infliximab every 8 weeks rather than every 4 weeks as recommended.\nMagnetic resonance (MR) angiography of the chest, abdomen, and pelvis and Cardiac MR three months later showed luminal narrowing of the distal thoracic and upper abdominal aorta similar to previous CT studies, stenosis of origin of celiac axis, stenosis of proximal superior mesenteric artery (SMA), and moderate stenosis of bilateral proximal renal arteries. Echocardiogram noted concentric LVH with mildly reduced function, measuring 45%, and normal coronary arteries.\nAt this point, the patient transferred her care to our rheumatology clinic, and despite treatment with infliximab 5 mg/kg every 8 weeks, she continued to report right thigh pain and developed new inflammatory arthritis of the left ankle and increased inflammatory markers with an ESR of 55 mm/hr (nl 0–20 mm/hr) and an elevated CRP of 73 mg/L (nl < 3 mg/L). Additionally, interval imaging found new wall thickening around the right common carotid artery. This was concerning for uncontrolled TA and CRMO activity. There was an unfortunate delay in treatment escalation due to social circumstances. Two months later, she started treatment with parenteral methylprednisolone 1 gram weekly for 8 weeks, and her dose of infliximab was increased from 5 mg/kg to 10 mg/kg every 4 weeks. Based on adult data demonstrating a positive response to higher doses of infliximab, the decision was made to increase the dose of infliximab instead of trying another TNF inhibitor.\nMultidisciplinary evaluation at Boston Children's Hospital, Center for Middle Aortic Syndrome by neurosurgery, nephrology, and rheumatology, led to recommendations of a prednisone dose increase and the addition of methotrexate 15 mg/m2 weekly to infliximab 10 mg/kg every 4 weeks. At this time, her echocardiogram revealed moderate left ventricle dilation and mildly depressed left ventricular systolic function. One month later, repeat head and neck CTA showed progression of her right carotid artery stenosis to 80%. Her ESR had normalized, and CRP decreased to 7.8 mg/L at this time. Given her worsening carotid artery stenosis, infliximab was increased from 10 to 15 mg/kg every 4 weeks. On a combination of moderate dose prednisone, weekly methotrexate, and infliximab, she denied joint pain, swelling, abdominal pain, diarrhea, or blood in stool. Follow-up brain MRI/MRA three months later was normal. Repeat chest and abdomen MRA showed all of the areas of stenosis appeared to be stable and inflammatory markers had normalized.\nCurrently, her clinical course is stable without further anatomic progression, and she has normal inflammatory markers. Thus, she has continued on her current regimen with infliximab 15 mg/kg every 4 weeks and methotrexate 15 mg/m2 once weekly. Prednisone decreased gradually and discontinued. She has not developed any infectious sequelae on this regimen. Her blood pressure is stable on carvedilol alone. We continue to assess blood work monthly. Given her use of prednisone, vitamin D level was followed and found to be low, and she has started on a vitamin D supplement. Dilated ophthalmologic exam was normal. Repeat MRI of the lower extremities and bone scan show that her CRMO lesions are inactive. Repeat MRI/MRA of the brain, chest, abdomen, and pelvis in June of 2018 show stable changes without the need for stenting. Given the predisposition for autoinflammatory conditions in this patient, the authors are considering genetic testing in search of a monogenic cause that may support a unifying diagnosis. |
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