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A 16-year-old male complained of inability to flex his left elbow since 1 year prior to admission. One and a half year before, he fell down and hit his elbow during football practice. He felt pain and there was swelling on his elbow. However, he didn't seek for medical treatment. He had his elbow massaged every week for 5 months but there was no improvement. His elbow became fixed in extended position. A month later, he went to an orthopaedic surgeon and underwent x-ray examination which revealed a fracture and dislocation on his left elbow. He was then referred to our institution for further treatment.\nFrom clinical examination, range of flexion-extension of the elbow was 300-00 with normal pronation-supination. There was no neurological deficit (). From radiological examination, there was a malunion of medial epicondyle with subluxation of left proximal ulna (). From 3D CT reconstruction, there was a deformity and malunion fracture in humeral capitellum with radial and ulnar postero-superior dislocation (). The patient was diagnosed with extension contracture of the left elbow due to malunion of left capitellum, neglected dislocation of the radiohumeral joint, and neglected dislocation of the ulnohumeral joint. The patient was scheduled to have a contracture release, open reduction and internal fixation, and ulnar interposition.\nIntraoperatively, we did a posterior approach to the elbow. The ulnar nerve was identified and preserved. The fibrotic tissues and heterotopic ossification were excised. We did a contracture release and open reduction and internal fixation using K-Wire. The flexion and extension of the elbow were evaluated and we managed to get 300 - 130° of flexion-extension ROM. Afterwards, ulnar interposition was performed to prevent ulnar impingement. The wound was closed and a single drain was placed. The elbow was immobilized with back-slab in 900 flexion position for two weeks.\nAfter 1 week, the patient went back to our hospital for follow-up examination. In the 1st evaluation, we tried to remove the back slab and moved the elbow passively. The movement is restricted due to pain and the patient went back home with the back slab on. In the 2nd week follow-up, we permanently remove the back slab and the stitches. At that time, the pain still persisted and the patient was planned to have physical rehabilitation.\nOn the 4th week after surgery, the surgical wound was infected. We performed debridement, implant removal, and manipulation under general anesthesia. Two weeks later, patient came back to our hospital. We removed the stitches and started rehabilitation. Later on, he continued his rehabilitation in his previous hospital.\nAfter 6 months, he visited our outpatient clinic for medical checkup. From physical examination, the elbow flexion-extension ROM was 1100 - 300 (). The patient is able to do normal daily activities (). |
A previously healthy 68-year-old woman awoke in the morning with dizziness which, by early evening, progressed to nausea, vomiting and a severe headache. Within 60 min of headache onset she became confused and somnolent. She had no history of hypertension, coagulopathy or illicit drug use. There was a history of low-dose aspirin use, which had reportedly been taken the day prior to the event. Upon initial presentation the patient's blood pressure was 178/70 mm Hg and pulse 72 bpm. A CT scan displayed intraparenchymal hemorrhages involving the left parietal and bilateral temporal lobes and a 3-mm rightward midline shift. Approximately 3 h after headache onset, the patient's blood pressure was 189/68 mm Hg, pulse 59 bpm and respiratory rate 9/min. MRI showed large areas of parenchymal hemorrhage in the right and left temporal lobes, the left parietal region and the right tentorium (fig. ). Small foci of restricted diffusion near the vertex and the posterior limb of the internal capsule were present. There was no abnormal enhancement following contrast administration. No source of hemorrhage was identified on cerebral angiography. Transthoracic echocardiography did not reveal valvular pathology. Blood cultures were negative. A brain biopsy was planned but not performed due to brain herniation and a worsening neurologic examination. The patient died on hospital day four.\nA complete autopsy was performed. Gross neuropathologic examination revealed intraparenchymal hemorrhage involving the left and right temporal lobes, softening of the medial temporal lobes secondary to transtentorial herniation and punctate hemorrhages in the superior portion of the left temporal lobe. Microscopic examination of the brain showed hemorrhagic infarctions within the bilateral temporal lobes, the right frontal and parietal lobes and the left occipital lobe. Thickened, hyalinized blood vessels were noted in the frontal, temporal, parietal and occipital lobes. Congo red stain and beta-amyloid immunohistochemical stains displayed multiple congophilic and beta-amyloid-reactive blood vessels (fig. ) as well as neuritic plaques. Blood vessels adjacent to regions of hemorrhage showed microangiopathic change, with fibrinoid necrosis in addition to hyalinosis of the vessel walls. While no definite parenchymal microhemorrhages were identified by imaging, occasional foci of perivascular hemosiderin deposition with spongiosis indicative of prior hemorrhage were found on microscopic examination. Vascular malformations, onion-skin-type thickening or lipohyalinosis characteristic of hypertensive microangiopathy and saccular aneurysms were not identified. There were not significant numbers of perivascular inflammatory cells, providing no support for vasculitis. Grocott methenamine silver stain and Gram stain were negative for microorganisms. PCR of brain tissue did not detect the presence of HSV-1 or HSV-2 nucleic acid. Culture of brain tissue and immunohistochemical staining for HSV-1 and HSV-2 were negative. |
A 59-year-old man was admitted to our hospital with symptomatic COVID-19 for 5 days. The diagnosis was based on a nasopharyngeal swab using the reverse transcription-polymerase chain reaction test for SARS-CoV-2 RNA and chest computed tomography (Fig. ). Two days after admission, that is, 7 days after symptom onset, his dyspnea worsened, and he required endotracheal intubation. He was subsequently placed on mechanical ventilation for 20 days. On the 30th hospital day, his nasopharyngeal swab PCR test result turned negative, and he was transferred from the intensive care unit to the general ward. The antibiotics and corticosteroids were stopped as the general condition improved. However, he required oxygen supplementation via nasal cannula with a flow of 2 L per minute of oxygen. The pulmonologist diagnosed the patient with pulmonary fibrosis and referred him to the rehabilitation department (Fig. ).\nAt the start point of rehabilitation, his motor grades were 5/5 in upper limbs and 4/4 in lower limbs. He could walk independently, but the endurance was limited to 200 m with oxygen supplementation. We decided to provide a conventional pulmonary rehabilitation program for pulmonary fibrosis according to “Consensus Document on Pulmonary Rehabilitation in Korea, 2015”.[\nThe program consisted of 10 supervised physical therapy sessions according to Frequency, Intensity, Time, and Type principles: 5 days a week of frequency, 12 to 14 on Borg Rating of Perceived Exertion (RPE) of intensity, 60 minutes per day of time, stretching, strengthening and aerobic exercises of type. To keep the exercise intensity to 12–14 of RPE, the physical therapist checked the RPE and managed the intensity every 10 minutes (see Supplementary Exercise protocol).\nAt the first session, he could not perform the 1-hour exercise program without oxygen supplementation. However, on the 8th day, he did not require oxygen supplementation during the whole exercise session. The functional evaluation showed improvements (Table ). Thus, he was discharged without oxygen supplementation on the 49th hospital day.\nThis case report confirms CARE case report guideline checklist (see Supplementary Checklist). Written informed consent was obtained from the patient for publication of the case details and accompanying images. |
A 66-year-old woman with a significant past medical history of well-controlled hypertension was admitted with complaints of microscopic hematuria and mild proteinuria for the past 3 years. Serum creatinine level was within normal range at that time and therefore the anti-GBM antibody was not tested. The first renal biopsy revealed mesangial proliferative glomerulonephritis with fibro-cellular crescents in one out of 18 glomeruli, excluding one global sclerotic glomerulus (Fig. ), and deposition of IgA and C3 in mesangial areas by immunofluorescence microscopy (Fig. ). Weak but significant IgG deposition was also observed in glomeruli in the distribution somewhat different from IgA or C3 (Fig. ). The electron-dense deposits were observed in mesangial areas by electron microscopy. Therefore, the diagnosis was IgA nephropathy. Antihypertensive therapy was initiated, mainly with an RAS inhibitor. Eight months later, the patient’s serum creatinine suddenly rose to 4.53 mg/dL (it was 1.04 mg/dL from the routine blood test 1 month before). Urinalysis showed 100 red blood cells per high power field and urinary protein excretion of 12.3 g/gCr (Fig. ). The serological tests that were performed to differentiate the cause of rapidly progressive glomerulonephritis revealed the presence of anti-GBM antibody at the titer of 116 IU/mL and the absence of anti-nuclear antibody and anti-neutrophil cytoplasmic antibody. Laboratory findings on admission are summarized in the Table .\nAfter admission, treatments with hemodialysis, plasma exchange, and intravenous methylprednisolone pulse therapy followed by oral prednisolone at the dose of 50 mg/day were initiated. The second renal biopsy was performed at 4 weeks after admission in order to assess the probability of renal recovery and to make the final diagnosis. It revealed cellular to fibrocellular crescents in 18 of 25 glomeruli, excluding six global sclerotic glomeruli by light microscopy. By immunofluorescence study, linear IgG deposition along the glomerular capillary walls and mesangial staining for IgA were observed. On the other hand, C3 deposition was observed in the mesangium as well as in the glomerular capillary walls (Fig. ). Electron-dense deposits were observed in mesangial areas, similarly as in the first biopsy, by electron microscopy (Fig. ). Based on the aforementioned findings, the diagnosis of anti-GBM glomerulonephritis and IgA nephropathy was confirmed. Plasmapheresis was performed eight times, anti-GBM antibody gradually decreased, and alveolar hemorrhage was prevented. However, her renal function could not be restored and she underwent maintenance hemodialysis (Fig. ).\nAdditional immunosuppressant was not given because the patient did not show any sign of pulmonary involvement and because the renal recovery was quite unlikely from clinical (continuous oliguria and hemodialysis dependence) as well as histological (crescent formation in most of non-sclerotic glomeruli) point of view.\nClinical and histological presentations from IgA nephropathy (at the time of first renal biopsy) and from anti-GBM disease (at the time of second renal biopsy) were summarized in the Table . |
A 28-year-old woman was admitted to the hospital with progressive ocular symptoms as well as newly developed hemianesthesia and mental signs. Half a month before hospitalization, she complained of double eyelid droop and diplopia; the ocular weakness and fatigue was stable throughout the day and had nothing to do with physical activity. The local hospital suspected myasthenia gravis, a computed tomography scan of her brain revealed nothing, and her serum was collected to detect the acetylcholine antibody. While waiting for a hospital bed, she developed numbness in her right upper and lower extremities. At the same time, she presented with prolonged sleep duration and mental symptoms. She was then transferred to our institution. On admission, she was noted to have difficulty walking by herself due to truncal ataxia. There was no cold, fever, or diarrhea before her symptom onset.\nShe had been suffering from chronic anemia, and her family reported that she had dietary deficiencies due to poor appetite although she denied it herself. She was on no other medications, not pregnant, and did not use alcohol or illicit drugs.\nOn physical examination, the patient presented obviously unbalanced nutrition; she was 1.65 m in height yet only weighed 40 kg. She was conscious and oriented and scored 23 in the MMSE, mainly due to impaired memory and calculation performance. Eye signs were obvious; she had drooping eyelids (both 5 mm). The ophthalmoplegia was remarkable; she demonstrated bilateral abducens nerve palsies and adduction deficit of both eyes. Limitation and nystagmus on vertical gaze were also noted. There was slight dysmetria on both finger–nose and heel–shin testing. Limb dysdiadochokinesia was also observed. Bilateral pyramidal signs were positive. Sensory examination indicated hyperesthesia on the right side. Findings on muscle tone and strength tests were normal.\nShe had an abnormal Hb level of 92 g/L, accompanied by low levels of serum folate and ferritin; vitamin B12 level was normal. Laboratory tests also revealed elevated levels of CA199 and CA242; both were gastrointestinal tumor markers although an abdominal enhanced CT scan reported no findings. The CSF revealed an increased white blood cell count of 28/mL (dominantly lymphocytes); other routine analyses of the cerebrospinal fluid (CSF) were normal, including protein level and IgG index (0.49). Also, no oligoclonal bands were found. Additional laboratory workup—including rheumatology analysis—was negative.\nMagnetic resonance imaging of the brain () showed symmetric T2 hyperintensities predominantly located within periaqueductal gray matter and periventricular regions of the fourth ventricles. A diffusion-weighted sequence also showed a hyperintense signal along the floor of the fourth ventricle. The presence of a lateral ventricle lesion was also noted. All brain lesions did not show contrast enhancement. Nerve conduction studies and visual evoked potentials were normal.\nIn view of the possible diagnosis of WE, thiamine was started intramuscularly (300 mg 12-h). However, poor clinical recovery was observed. Her mental status worsened and exhibited hallucination, and the ocular palsy also progressed.\nThen came the crucial result that her serum and cerebral fluid tested positive for NMOSD-IgG (1:32 and 1:3.2, respectively). Thereafter, the spinal cord was evaluated, and MRI of the spine revealed a suspicious enhanced T2-weighted signal at the lower thoracic cord region ().\nWe corrected the diagnosis, and the patient started with treatment of high-dose intravenous steroids, but still little improvement was detected. The patient then experienced bilateral visual loss, but MRI results of the optic nerve reported no findings. After that, intravenous immunoglobulin treatment was given, followed by intravenous cyclophosphamide therapy. On discharge, she showed moderate improvement in eye movement and ataxia although her vision remained unchanged. |
A 21-year-old male presented to orthopedic outpatient department with a history of the left hip pain for 1 year, following a trivial fall before 1 year. The patient was not able to do heavy strenuous activities such as running, jumping, and other sports activities. Terminal range of movements were painful. FABER test was positive. The patient was able to raise his leg actively. Radiograph left hip revealed a radiolucent swelling along inferior part of neck with a well-defined, eccentric osteolytic lesion involving subcapital region of the left femoral neck. The lesion was oval shaped and without any calcification or periosteal reaction ().\nMagnetic resonance imaging (MRI) suggested of cystic lesion involving synovial lining near head-and-neck junction of the left femur ( and ).\nUnder regional anesthesia, with patient in supine position, the lesion was approached through smith and Peterson approach to the left hip. After cutting the left hip capsule in T shape and pulling the hip in flexion abduction and external rotation, the lesion was found. It contained rubbery and gelatinous material along synovial lining of inferomedial capsule with slight erosion of inferior neck. Curettage of the lesion was done. The bone defect was not found to be large enough to be filled with bone graft. Histopathological examination showed lobular pattern with stellate to spindle-shaped cells on the myxoid background (). Few osteoclast type of giant cells were found in the periphery of the lobules. Tumor cells had oval- to spindle-shaped nucleus with surrounding densely eosinophilic cytoplasm within a myxoid background. It was consisted with fibromyxoid chondroma.\nIn post-operative period, isometric quadriceps exercises were started on the same day of surgery. Knee and ankle range of movement exercises were started since the next day of surgery. He was discharged after 2 days with non-weight-bearing walking with walker support. He was followed at 4 weeks, 8 weeks, and 3 months. The patient was allowed on full weight-bearing from 2 months. He has a painless gait with no complications or recurrence till date. |
A 1-day old female newborn presented with severe unilateral proptosis (Fig. a, b). She was born at term with elective caesarian section after an uncomplicated pregnancy. The 25-year-old Caucasian mother had a normal antenatal history, no history of recreational drug use, no pharmacologic therapy or known exposure to chemicals or radiation.\nOphthalmologic examination revealed massive proptosis of the right eye and an absent direct or consensual pupil reaction of the right eye. Despite the degree of proptosis, possibly due to the very early examination of the newborn immediately postpartum, the cornea was clear. The anterior segment was examined via a portable slit lamp and showed neovascularization of the iris without hyphema. The lens was clear and there was a good red reflex of the posterior segment.\nA CT of the brain and orbits showed a large heterogeneous mass arising entirely in the right orbit, engulfing the globe and the optic nerve, and filling the entire orbit. No bony erosion or calcifications were noted (Fig. a). T2-weighted MRI imaging shows predominantly the tumor to be hyperintense to fat and extraocular muscles (Fig. b).\nDue to the degree of proptosis and the imminent risk for severe exposure keratopathy and corneal breakdown, urgent surgical excision of the tumor was scheduled. One day later, the tumor was removed completely via a 360-degree conjunctival limbal peritomy, identification of the 4 recti muscles and temporary disinsertion of the lateral rectus muscle in order to provide the necessary dissection plane to allow for complete tumor excision (Fig. a).\nAfter tumor removal there was significant upper and lower eyelid laxity and conjunctival prolapse due to an expansion effect on the eyelid and periocular tissues exerted by the tumor (Fig. b). However, it was decided not to sacrifice eyelid or conjunctiva tissue at this time and a decision to place upper and lower forniceal sutures and a reversible suture tarsorrhaphy and allow the tissues to retract to their normal size was made, (Fig. c). Since the prognosis for vision in that eye was very poor, it was decided to keep the tarsorrhaphy sutures for 3 weeks in order to help with the healing process and keep the “redundant” and laxed tissues in place. The eyelid tone and the conjunctival fornices were restored during this time. The tumor measured 7X5X4.5 cm (Fig. d).\nThe specimen was stained using a hematoxylin and eosin dye. Figure a shows the mature teratoma containing cells coming from three of the intestinal wall’s cell layers, namely the smooth muscle layer, the submucosa, and the mucosa with its columnar epithelium, while in Fig. b another portion of the specimen shows cells forming a choroid plexus papilloma. Figure c depicts respiratory epithelial cells alongside bronchial mucosal glands as well as bronchial cartilage tissue.\nPostoperatively, our newborn patient recovered remarkably well. Unfortunately, as expected due to the compression effect and to the ischemic changes manifested by the neovascularization of the iris, there was no vision from that eye, but the orbit developed remarkably well over the next years. We decided to follow the child clinically over the next 18 years and to proceed with orbital imaging only if required by the clinical exam findings. Our decision was based on complete tumor removal with clear margins, on the very low malignant potential and on the requirement for general anesthesia during imaging. The anterior segment hypoxia reversed as shown from the absence of iris neovascularization. There was a progressive mild to moderate volume deficit of the right orbit manifesting mainly as a deepened superior sulcus and a corresponding upper eyelid ptosis/contour abnormality (Fig. a, b). When our patient was mature enough to wear a scleral shell, a custom-made shell was fitted. The family elected not to undergo any further surgery or minimally invasive procedures for cosmetic reasons at this point. Figure a and b shows our patient currently at age 18.\nThere was no recurrence or malignant transformation during this 18-year follow-up period and our patient graduated from high school and was recently admitted to Law school. |
We present a case of 21-year-old female patient with a negative medical and surgical history. She presented to the gastroenterology clinic for chronic epigastric pain for several months, associated with postprandial episodes of nausea and vomiting. Her physical examination was insignificant. She was prescribed proton pump inhibitors and prokinetics.\nHowever, her symptoms didn't improve much, so a diagnostic upper esophagogastroduodenoscopy was done and showed normal mucosa but with external compression on the second part of the duodenum (\n). Computed tomography (CT) scan of the abdomen and pelvis was ordered due to characterize the external compressing mass. It showed a diverticulum at the second part of the duodenum (D2) measuring approximately 41 mm × 28 mm, filling the duodenal lumen (intraluminal diverticulum) and obstructing it (\n). Finally, an endoscopic ultrasound was done to delineate the diverticulum and have a clearer view of the surrounding structures. It showed again the intraluminal duodenal diverticulum (IDD) in D2, with a thin and regular wall. The endoscopic ultrasound also confirmed normal pancreatic appearance and normal Wirsung's duct and biliary tract.\nThe patient was presented with her options and after a surgical consult, and due to her intractable symptoms, it was decided to go with surgical treatment. Written consent was taken from the patient. She was prepared for the operating room.\nIntraoperatively, the duodenum was first completely exposed. Then, an intraluminal mass was palpated at the level of D2. Through a longitudinal duodenotomy, the intraluminal diverticulum was identified. The diverticulum was identified 20-mm proximal to the papilla. The papilla was intubated to avoid injury during traction of the mucosa. Excision of the diverticulum was then performed and the mucosa was approximated using absorbable sutures.\nThe postoperative course was unremarkable and the patient was discharged on day 6 after regaining bowel movement and tolerating oral intake. During her follow-up in the outpatient department, a week after her discharge, she reported complete resolution of her preoperative debilitating symptoms of pain and nausea. |
A 64-year-old woman was referred to correct the prosthodontic restoration on the right maxillary first incisor with infraocclusion. This patient had been treated by implant therapy 20 years before. She was in good general health and did not present any contraindication to surgery procedure, but she had a high tobacco consumption, which she had decided to reduce. Clinical examination showed chronic periodontitis, the replacement of the right maxillary first incisor by an implant with infraocclusion, and a crown with a slight palatal position (Fig. ). Soft and hard tissues around the implant and osseointegration were stable. The facial skeletal pattern was normodivergent. The actual implant position was due to 20 years of continuous alveolar growth. Considering the good palatal implant positioning at the time of the surgery, 20 years before, the right axis was kept, without any buccal side effect movement. A new screwed restoration would have been possible, but the prosthetic rehabilitation to compensate for the infraocclusion would have led to a disappointing result, with too long a crown and an incorrect gingival line alignment. For this reason, periodontal treatment was planned first before orthodontic preparation and implant relocation at the right level using the OBS technique.\nOrthodontic preparation consisted of teeth alignment excluding implant and a .021x.025 SSW was placed on the maxillary and mandibular arches before surgery. When the alignment phase was completed, infraocclusion on the incisor prosthodontic crown was severely worsened, compared to the incisal edge of the adjacent teeth (Fig. A). Immediately before surgery, an overlay .016 Nickel-Titanium archwire was placed on maxillary teeth, including the implant. (Fig. ).\nThe OBS procedure was performed on the buccal side with preservation of the implant soft tissue attachment. Immediately after surgery, orthodontic forces were applied. Every 2 weeks (Fig. ), the patient was followed by the orthodontist for traction reactivation. After 2 months of orthodontic traction, the movement was stopped. New surgery was planned on the palatal side. Immediately after surgery, orthodontic traction was applied along the desired axis. Bone stretching movement allowed implant extrusion. After stabilization, a new crown was made and a retainer was bonded (Fig. ). |
A 53-year-old man presenting with chronic dysphasia and chest pain was admitted to our hospital. He had been treated with an unidentified complaint by a physician. Upon admission, his blood pressure was 128/62 mmHg symmetrically. His pulse was regular (72 beats/min). Neither carotid bruit nor cardiac murmur was reported. Lung examination revealed clear breath sounds. Laboratory evaluations did not show any abnormalities. Transthoracic echocardiography (TTE) revealed normal left ventricular function unassociated with congenital heart disease. Computed tomography angiography (CTA) demonstrated an RAA and an ALSA originating from a KD (). Cross-sectional mesurements of the KD revealed that the diameter of the diverticulum from the opposite aortic wall to the tip of that was 52 mm, and the diameter at the origin of the ALSA from the aortic arch was 27 mm. The aortic arch vessels arose in the following order: left common carotid artery, right common carotid artery, right subclavian artery, and ALSA. The fourth branch posteriorly passed to the esophagus from a bulbous diverticulum across the midline toward the left and upward. The left vertebral artery arose from the left common carotid artery. The patient was considered to be unsuitable for thoracic endovascular aortic repair (TEVAR) alone because of endoleak with insufficient proximal landing zone of stent-graft. We decided to perform two-stage surgery with endovascular treatment after written informed consent was obtained.\nTo ensure revascularization of the bilateral upper extremities, the bilateral axillary arteries were first prepared to be anastomosed with an 8-mm ringed polytetrafluoroethylene graft. Through a median sternotomy, cardio-pulmonary bypass was instituted in standard fashion with ascending aortic and bicaval cannulation. The KD and an adjoining ALSA in the surgical field was difficult to confirm. Under deep hypothermic circulatory arrest, both common carotid arteries were reconstructed using the arch-first technique with retrograde cerebral perfusion. The orifice of the right subclavian artery was doubly ligated. Antegrade cerebral perfusion was started through a four-branch Dacron graft. Distal anastomosis, located on the right side of the trachea, was created just behind the right common carotid artery without an elephant trunk. Subsequently, the tube graft anastomosed at the distal side was combined with the main graft to achieve cerebral perfusion using a stepwise technique. Resumption of the whole body was performed, and proximal anastomosis was achieved with the transected aorta just above the sino-tubular junction. According to the reconstruction between graft conduits anastomosed to the bilateral axillary arteries and the side branches of the four-branch Dacron graft retrieved through the thorax, total arch replacement was completed. The postoperative hemodynamics state was stable. The artificial conduit competed with the native left subclavian artery with regard to blood flow. Because there was a concern that the graft anastomosed to the left axillary artery might be occluded by a thrombus, an endovascular procedure was performed the next day. The time interval between prior total arch replacement and TEVAR was 16 h. The right femoral artery was exposed through an oblique incision. A 31×150-mm conformable GORE TAG stent-graft (W. L. Gore and Associates, Inc., Flagstaff, AZ, USA) was advanced in retrograde fashion across the angulated aortic arch and subsequently deployed just distal to the side branch of the four-branch Dacron graft that controlled the perfusion to the right arm. Because endoleak was detected using angiography, the 31×100-mm conformable GORE TAG stent-graft was additionally inserted to ensure the coverage of the ostium of the KD. The left brachial artery was percutaneously cannulated with a 6Fr introducer (Destination, Terumo, Tokyo, Japan). For the purpose of resolving dysphagia, we did not select means to completely occupy the KD by coils. To completely exclude the KD, the non-dilated origin of the ALSA was intentionally embolized with detachable coils (MICRUSFRAME; Johnson & Johnson, New Brunswick, NJ, USA) to accurately construct the frame at an early stage. Further, the space in the frame was packed with pushable coils (Tornade Embolization Coil; Cook, Bloomington, IN, USA) to reinforce the embolic effect. The completion angiography demonstrated a complete exclusion of the diverticulum. The postoperative course was uneventful without cerebrovascular failure, respiratory failure, or paraplegia. Postoperative CTA demonstrated good patency of the bypass grafts to the supra-aortic vessels, and complete exclusion of the KD by the stent-grafts and coils (). The patient’s dysphagia resolved, and he is being followed up for medical treatment once a month after discharge from the hospital. |
An 80-year-old woman presented with a history of intermittent, severe epigastric pain. Over the preceding 5 months, she had less severe and self-resolving epigastric pain 15–30 min after every meal. The full blood count, serum biochemistry values, and C-reactive protein level were within normal ranges. Additionally, arterial gas analysis was normal. Abdominal plain radiography showed free air between the liver and diaphragm, and PI (Fig. ). Abdominal computed tomography showed severe calcification of the SMA origin; SMA peripheral flow was reserved (Fig. ). Computed tomography (CT) revealed bubble-like intramural gas of the small bowel with the contrasted wall pneumoperitoneum (Fig. ).\nSince the patient did not have peritonitis, a conservative approach was performed. She was managed in the condition of intensive care unit, due to fears of the potential for acute mesenteric ischemia due to mesenteric artery occlusion or non-occlusive mesenteric ischemia. She was managed for bowel obstruction, which included fasting and intravenous fluid administration. She received heparin infusion to prevent SMA occlusion and maintain collateral flow. During admission, she reported abdominal pain relief. Seven days after admission, abdominal plain radiography showed improvement in PI and pneumoperitoneum; therefore, she was permitted to begin drinking fluids. There was no evidence of recurrent abdominal pain after the fluid consumption, so she was allowed to eat solid food.\nAngiography was planned to relieve the postprandial abdominal pain. The findings showed severe stenosis of the SMA origin with calcification, and the SMA had replaced the common hepatic artery (Fig. , ). ET, namely stenting to the SMA and common hepatic artery, was performed. The SMA trunk was engaged with a 6F Parent Plus 60 guiding catheter (Medikit, Tokyo, Japan) from the left brachial artery. Initially, 8000 units of heparin was infused, and additional heparin was added to keep the activated whole blood clotting time over 200 s. The SMA occlusion was traversed using a 0.014” NEO EVT Guide Wire Cruise (ASAHI INTECC J-sales, Tokyo, Japan). The SMA trunk to the hepatic artery was traversed using a 0.014” NEO EVT Guide Wire Cruise, which was engaged with a 6F SheathLess PV (Cardian Health, Ohio, USA) from the right brachial artery. Intravascular ultrasonography (IVUS) (Navifocus WR, TERUMO, Tokyo, Japan) revealed severe stenosis of the SMA trunk with calcification. Balloon dilation was performed with the kissing ballon technique using a 4-mm Coyote ES (Boston Scientific, Marlborough, MA, USA) to the SMA and 5-mm SHIDEN RX (KANEKA MEDICAL, Osaka, Japan) to the common hepatic artery. After dilation, two balloon-expandable stents (5-mm PALMAZ Genesis (Cardian Health, Ohio, USA) to the SMA and 6-mm PALMAZ Genesis to the common hepatic artery) were implanted with the kissing stent technique from the common ostium (Fig. ). The sizes of the stents were based on the IVUS measurements. IVUS showed good orifice expansion. Final angiography showed satisfactory circulation to the terminal organ from the orifice (Fig. ).\nEndovascular therapy relieved the patient’s symptoms. She required dual anti-platelet therapy (DAPT) with aspirin and clopidogrel post-intervention. Follow-up computed tomography showed patency of the orifice of the common hepatic artery and SMA, and there was no evidence of PI or pneumoperitoneum. No recurrent postprandial abdominal pain occurred for two years after the ET. |
We describe the case of a 35-year-old white woman. She initially presented in July 2012 at the age of 27 years, when she was working as a nurse. She reported subacute onset of lower limb weakness (strength 3/5 on the Medical Research Council scale), lower limb numbness, gait imbalance, and urinary incontinence. Magnetic resonance imaging (MRI) of the thoracic spine, with and without contrast, demonstrated signal abnormality from T7 to T10 associated with post-contrast enhancement. MRI of the brain and cervical spine, with and without contrast, was normal. Visual evoked potential (pattern VEP) showed normal P100 latencies. A lumbar puncture showed cerebrospinal fluid pleocytosis (white blood cells: 20 cells/dL, with predominant lymphocytes), normal IgG index, and oligoclonal bands.\nA summary of treatment regimens and timelines is shown in and the clinical course is described in . Following her initial presentation the patient was admitted to hospital and treated with intravenous methylprednisolone at a dose of 1 g/day for 5 days, after which she was discharged without medication to acute rehabilitation. She had a relapse in October 2012 and underwent PLEX every other day for a total of five exchanges which resolved the episode; a relapse was defined as patient-reported symptoms or any new signs consistent with CNS lesions and attributable objective changes in MRI or visual evoked potential. The patient was treated with intravenous methylprednisolone for relapses between 2012 and 2014, during which she experienced multiple episodes of lower limb weakness, urinary incontinence, and falls. During these relapses, MRI of the thoracic spine showed contrast enhancement from T6 to T10. In 2014, the patient tested negative for AQP-4 IgG and in February she was initiated on oral prednisone 40 mg/day. Her Expanded Disability Status Scale (EDSS) score was 4 at that time.\nIn 2015, testing for anti-AQP-4 antibodies by fluorescence-activated cell sorting (FACS) proved negative. In March of that year, the patient's oral prednisone dose was reduced to 20 mg/day because of weight gain. In June 2015, the patient was initiated on mycophenolate mofetil 1 g twice daily.\nIn 2016 and 2017, testing for MOG IgG (FACS assay) was negative; tests for antinuclear antibody panel, urine heavy metal screen, and paraneoplastic panel were also negative. In February 2017, following episodes of intractable nausea and vomiting with associated area postrema changes on MRI, the patient received a diagnosis of seronegative NMOSD from a National Multiple Sclerosis Society Center of Excellence, based on recurrent episodes of LETM, MRI changes, area postrema syndrome, and lack of anti-AQP-4 antibodies. The patient continued to experience relapses between 2014 and 2019, with 4–5 relapses per year (), characterized by lower limb weakness. On average, the patient was hospitalized three times a year for treatment of relapses with intravenous methylprednisolone 1 g/day for 5 days, with maintenance PLEX applied every 4 weeks from March 2018 to April 2019 as a total of 3–5 exchanges on alternate days. In June 2018, the patient stopped mycophenolate mofetil treatment as she felt it was not effective. Between 2014 and 2018, the patient's EDSS score was 4–5, and she eventually needed a cane to walk in 2018.\nStarting in July 2018, rituximab 375 mg/m2 was administered intravenously every week for 4 weeks, then as two 1 g doses, 2 weeks apart every 6 months. Rituximab has not been approved for treatment of NMOSD; this regimen is the same as that used in a Phase 3 trial in patients with NMOSD (). In August 2018, the patient stopped oral prednisone treatment [due to weight gain of 20 lb (~9 kg) in 2 months, acne, and mood issues]. Rituximab treatment was stopped after the second maintenance cycle was completed on July 8, 2019, as no significant improvement in relapses occurred—the patient experienced three relapses while receiving rituximab (not confirmed by MRI). CD19+ B cells were depleted during rituximab therapy, with the median cell count for the last 6 months of treatment being 0.001 × 109cells/L (range 0–0.0159 × 109 cells/L). Eculizumab infusion was initiated on July 30, 2019, with the approval of the patient's insurance provider following a peer-to-peer review by one of her physicians (RG) and the provider. Two weeks before eculizumab initiation, the patient was vaccinated against Neisseria meningitidis with meningitis ACWY and B vaccines, according to the recommendations of the Centers for Disease Control and Prevention's Advisory Committee on Immunization Practices (). She then received the recommended dose of eculizumab 900 mg weekly for the first four doses, followed by 1,200 mg every 2 weeks starting 4 weeks after initiation. While treated with eculizumab, the patient showed improvements on the EDSS (score of 2–3; lower scores indicate less disability) () and she experienced no relapses or adverse events. Eculizumab was discontinued in December 2019 when the patient's insurance provider denied continued coverage despite peer-to-peer review. At subsequent follow-up visits after eculizumab discontinuation and as of August 2020 she has remained relapse-free and symptom-free and is not taking any medication for NMOSD. The patient can walk without any aids, has an EDSS score of 2, and works full time as a physician's assistant. |
A 66-year-old female, with a past medical history for hypertension, presented to hospital with a 12-hour history of an acute onset severe headache and neck pain. Prior to arrival at hospital, she had progressive deterioration in her level of consciousness (LOC). Upon arrival to the emergency department, she was with GCS of 9 (motor = 5, eyes = 2, and verbal = 2). There was no witnessed seizure activity, and the patient was hemodynamically stable throughout transport. No episodes of hypotension or hypoxemia were recorded. She had a definitive airway obtained via endotracheal intubation and was sent for computed tomography (CT) of the brain.\nThe CT of the brain displayed a Fisher grade 4 SAH with significant clot burden in the interpeduncular, carotid, and ambient cisterns bilaterally (Figures and ). In addition, the imaging displayed hydrocephalus. Computed tomographic angiography (CTA) of the circle of Willis (COW) was obtained and failed to display a discrete source for the SAH. Three-dimensional reconstructions of the CTA were unclear but displayed a questionable small aneurysm in the area of the left anterior choroidal artery.\nGiven the Fisher CT grade 4, Hunt and Hess clinical grade 4 (with a GCS of 9 and stuporous), and the World Federation of Neurological Surgeons (WFNS) clinical grade 4 SAH with the presence of hydrocephalus, a right frontal external ventricular drain (EVD) was placed. The EVD was left open at 20 cm above the tragus. The patient was transferred to the intensive care unit (ICU) with the plan for a 4-vessel digital subtraction angiogram (DSA), in order to better delineate the suspected anterior choroidal artery aneurysm. The patient improved to a GCS of 9T (motor = 6, eyes = 3, and verbal = T) after EVD placement.\nThe DSA images confirmed the presence of the aneurysm (Figures and ), and the patient was taken to the operating room for microsurgical clipping 6 hours after admission to hospital.\nIntraoperatively the procedure went without complication, with the exception of the need to sacrifice one large sylvian vein. Postoperative CT of brain displayed small subfrontal hypodensity consistent with venous infarct. The patient was clinically unaffected by this.\nPostoperatively the patient returned to the ICU where she remained ventilator dependent on pressure support of 20 with a positive end expiratory pressure (PEEP) of 8 cm H2O and a fractional inspired oxygen (FiO2) requirement of 50%. Chest X-ray displayed pulmonary edema. She had mild tropinemia, with no significant electrocardiogram (ECG) changes. A transthoracic echocardiogram displayed biventricular failure (right worse than left) and global hypokinesis, with an ejection fraction (EF) of 40%, all consistent with catecholamine related subendocardial ischemia. She remained on minimal sedation for comfort during her ICU stay, with a fentanyl infusion at 25 mcg/hr. This was not changed at any point during her ICU admission. Nimodipine was administered only once, at a dose of 60 mg, and resulted in a decrease in MABP from 85 mm Hg to 55 mm Hg. Given this response to nimodipine and the echo results, it was elected to not administer any further doses.\nOver the following 5 days after bleed, she remained ventilator dependent secondary to her pulmonary edema. We consistently recorded central venous pressure (CVP) between 12 and 15 mm Hg. Mean arterial blood pressure (MABP) was maintained spontaneously between 80 and 90 mm Hg.\nOn postbleed day 5, she had a brief episode of right arm weakness lasting for 30 seconds, with return to baseline. There was no documented loss of consciousness. She remained able to obey commands both during and after this brief event. Uninfused CT of the brain failed to display any new abnormality. Electroencephalogram was conducted, with no seizure activity noted. Subsequently magnetic resonance imaging (MRI) of the brain was completed displaying signs of moderate to severe angiographic vasospasm of the basilar trunk, bilateral supraclinoid internal carotid arteries (ICA), and bilateral M1 segments of the middle cerebral artery (MCA) via time of flight (TOF) imaging (). Given only the presence of angiographic cerebral vasospasm via TOF MRI in the absence of clinical manifestations, it was elected to monitor the patient clinically.\nOn postbleed day 6, the patient became drowsy and unresponsive in the early afternoon. No changes in her hemodynamics were noted during this time, with MABP recorded between 80 and 90 mm Hg. An urgent CTA of the COW was ordered confirming the results from the MRI-TOF obtained the previous evening (). Given the significant proximal vasospasm, it was elected to arrange for angioplasty while the patient was optimized medically for the presumed diagnosis of symptomatic DCI secondary to cerebral vasospasm.\nIn the presence of significant subendocardial ischemia and pulmonary edema requiring ventilatory support, it was elected to avoid hypertensive therapy and trial of a high dose PDEI based therapy with IV milrinone following the previously described protocol from the Montreal Neurological Institute []. A 5 mg IV milrinone load over 10 minutes was given (0.1 mg/kg load; weight 55 kg), followed by continuous IV infusion of 0.75 mcg/kg/min. No hypotension or arrhythmia was encountered. The MABP during and after load was maintained at 80 to 95 mm Hg spontaneously.\nWithin 30 minutes of the IV milrinone load, the patient began to open her eyes spontaneously. Within 40 minutes of the infusion, the patient began to intermittently follow commands with all extremities. The patient was subsequently taken to the angiography suite for planned angioplasty approximately 1.5 hours after IV milrinone bolus.\nThe DSA prior to the planned angioplasty displayed significant angiographic resolution of the vasospasm in the basilar and bilateral ICA/MCA territories. Some mild residual spasm was noted at the basilar apex and the right proximal middle cerebral artery (). Given the improvement and lack of obvious target of angioplasty, the procedure was aborted and the patient returned to the ICU. It was noted at this time that the patient was back to baseline neurologically.\nThe patient was maintained on 0.75 mcg/kg/min of IV milrinone for 72 hours, after which we tapered the infusion by 0.25 mcg/kg/min every 24 hours. No complications of the milrinone therapy were encountered. The MABP was maintained spontaneously between 85 and 95 mm Hg while being on the IV milrinone therapy. We were also subsequently able to liberate the patient from the ventilator 2 days after the attempted angioplasty procedure.\nContinued improvement occurred during the following 1.5-week ward stay, with an eventual transfer to our stroke rehabilitation unit for ongoing therapy. |
A five-year-old male patient who had a two- month-old history of inability to bear weight with the left lower extremity presented to our institution’s pediatric oncology department in August 2016. Prior to his visit, imaging with X-rays and magnetic resonance imaging (MRI) followed by a biopsy were performed at an external center with a diagnosis of osteosarcoma (Figure 1). After presentation, his images and biopsy specimens were reassessed at our institution (per routine hospital policy) and the patient was diagnosed with Ewing sarcoma of the proximal femur. The mass was located at the intertrochanteric and subtrochanteric regions, accompanied by a minimally displaced pathological fracture at the femoral neck. Multiple metastatic nodules in his lungs were demonstrated on chest computed tomography (CT). The patient received six cures of vincristine, ifosfamide, doxorubicin, and etoposide (VIDE) as induction chemotherapy.\nAfter six cures of VIDE, the control MRI showed that the tumor shrank in response to chemotherapy, and was confined to the intertrochanteric region. With the femoral head epiphysis and the distal two-thirds of the shaft clear of tumoral involvement and the patient’s growth potential in mind, we deemed the patient a good candidate for both joint and epiphysis-sparing biological reconstruction. A written informed consent was obtained from each parent.\nWith the patient lying supine under general anesthesia, an incision was made from the anterior superior iliac spine to proximal femur. The previous biopsy tract was excised. After exposure, the femoral shaft was cut with a Gigli saw. The proximal femur was exposed, and the hip capsule was opened. The femoral head was cut from the physeal line. The shaft was cut 100 mm distal to the greater trochanter and the proximal femur was resected en bloc. The tumoral tissues were excised from the removed bone fragment. The remaining bone fragment was soaked in liquid nitrogen (LN) for 20 min. The cryoablated bone was left thawing for 15 min in room temperature and, then, kept in 30°C distilled water for 15 min.\nThe surgical field was closed with wet sponges. Afterwards, an incision was made at the left fibula. After exposure, a 120-mm metadiaphyseal segment of the proximal fibula was prepared as a vascularized autograft. The defect in the proximal femur metaphysis was reconstructed with the vascularized fibula covered by the LN-treated bone. The autografts were fixed to the femoral head epiphysis and shaft with a PHILOS plate (DePuy Synthes, Warsaw, IN, USA) and a headless compression screw (Artfiks, Ankara, Turkey) (Figure 2). The vascularized fibula was microsurgically anastomosed to the perforating femoral arteries and veins. A surgical drain was placed, and the wounds were closed. The patient was, then, transferred to the postoperative intensive care unit (ICU) as planned preoperatively.\nThe patient had an uncomplicated stay at the ICU and was transferred to the orthopedics inpatient clinic one day postoperatively. The graft’s viability was confirmed with a single-photon emission CT scan the day after surgery. According to the pathology report, the tumor had a 90% necrosis rate and surgical margins were clear. He was discharged with a unilateral hip spica cast (Figure 3a).\nThe patient lives abroad with his family and, therefore, adherence to our routine follow-up protocol was suboptimal. He received postoperative adjuvant chemotherapy in his home country. In the outpatient visit to our clinic nine months later, the patient began bearing full weight.\nThree years after surgery, the patient remained disease-free (Figure 3c), could walk without assistance, and was able to flex his left hip to 145° (Figure 4). He could manage most of his daily activities without assistance and socialize with his peers. His lower extremity Musculoskeletal Tumor Society (MSTS) score was 83% (25/30). On imaging, the proximal femur remodeled well and there was complete union. The femoral shaft had mild lateral bowing, and a shortness of 2 cm. The patient did not experience any serious complications; however, the headless screw pulled out in the postoperative ninth month and it was surgically removed. |
A 36-year-old Caucasian male presented to the emergency room complaining of a one-day history of abdominal pain. His main symptoms were that of nausea and vomiting, but he also reported periods of diarrhea. In the emergency room, his initial evaluation was significant for lab studies demonstrating a mild metabolic acidosis with a bicarb of 16.6 mEq/L, an elevation of his creatinine to 1.93 mg/dL, and a serum lactate level of 5.4 mmol/L. A computed tomography (CT) scan of the abdomen and pelvis was obtained and reviewed. The appendix was thought to be normal. There was no evidence of free intraperitoneal air, abscess, or volvulus. There did appear to be evidence of enterocolitis involving the ilium, cecum, and the proximal ascending colon. There was no evidence of pneumatosis or obstruction. Over the course of 12 hours the patient underwent conservative medical management, which included intravenous hydration, intravenous steroids, Toradol and Dilaudid for pain management, and the occasional dose of Ativan for agitation. Failure of conservative management along with medical decline lead to an exploratory laparotomy with a right hemicolectomy for an ileocolic intestinal infarction. Postoperative repeat CT scan of the abdomen incidentally showed pulmonary nodules in the lower lobe. A CT scan of the chest was then obtained, which revealed air in the left chest wall and axilla (Figure ). On physical exam there was very subtle mottling of the left shoulder. The patient was emergently taken back to the OR for further exploration of the shoulder with excisional debridement. Seropurulent fluid and extensive subcutaneous emphysema along the fascial planes of the left chest wall were consistent with necrotizing fasciitis. Both wounds were packed with a gauze bandage roll soaked in saline and then the patient was transferred back to the intensive care unit.\nOver the course of 15 days the patient underwent repeat trips to the OR for re-exploration, incision and drainage of the wounds, and dressing changes (Table ). On postoperative day 17, the wound, ostomy, continence nurse (WOCN) was consulted to evaluate the wound and make treatment recommendations.\nOn exam, there was still a significant amount of nonviable tissue in the base of the left chest and axilla wound. The left chest wound measured 8 x 19 x 8 cm and the left axilla wound measured 6.5 x 25 x 4 cm. NPWTi-d with ROCF-CC was applied to these wounds to aid in the removal of the nonviable tissue. Figures - show the presentation of the wounds prior to the application of the NPWTi-d with ROCF-CC.\nDuring the application of the NPWTi-d with ROCF-CC, care was taken to ensure that the contact layer of the dressing was in contact with the entire wound base. Barrier rings were applied to the skin creases along with any area that presented as a high risk for inadequate seal of the drape (Figure ). The therapy settings for the left axilla were 85 ml of hypochlorous acid solution (Vashe®, SteadMed, Fort Worth, TX), dwell time of 10 minutes with NPWT every hour at -125 mmHg. Therapy settings for the left chest wound were 50 ml of hypochlorous acid solution, dwell time of 10 minutes with NPWT every hour at -125 mmHg.\nDuring the first dressing change after the application of the NPWTi-d with ROCF-CC, there were notable improvements in the quality of the tissue in the wound bed (Figure ). The left chest wound bed was noted to have 100% red healthy tissue and the left axilla wound bed had a decrease in nonviable tissue. There was, however, seropurulent drainage noted deep within the intramuscular structures of the left axilla wound.\nNPWTi-d with ROCF-CC was reapplied to the axilla wound ensuring that the contact layer was placed deep into the wound bed to facilitate the removal of the seropurulent drainage. The left chest wound was transitioned to NPWTi-d to continue to promote granulation tissue (Figure ). The instillation solution was switched to normal saline due to blockage alarms we encountered with the hypochlorous acid solution without resolution.\nThe current treatment regimen was continued for two days until the next dressing change. At that time both wound bases were 100% red with granulation tissue present (Figure ). The seropurulent drainage deep in the intramuscular structures was no longer present. Since there was adequate removal of nonviable tissue and the quality of the tissue had significantly improved, the WOCN made the decision to transition both wounds to NPWTi-d instilling normal saline solution with a dwell time of 10 minutes and NPWT every two hours at -125 mmHg.\nFor the next two weeks the treatment regimen utilizing NPWTi-d to the wounds continued. The dressings were changed three times a week at the bedside by the WOCN. During initial assessment, the axilla wound measured in total volume 650 cm³ and the chest wound measured 1,216 cm³. After 17 days utilizing the combination of NPWTi-d with and without ROCF-CC, the axilla wound measured in total volume 342.25 cm³ and the chest wound measured 554.4 cm³. This presents a significant decrease in overall measurements. At this time, NPWTi-d was discontinued and standard NPWT with black granufoam was utilized to both wounds at -125 mmHg (Figures -).\nNPWT was utilized for approximately a month to promote granulation tissue and assist with wound contraction (Figures -). Once the wound had made vast improvements, NPWT was discontinued and a non-adherent antimicrobial alginate dressing (Silvercel™ Non-adherent, Acelity, San Antonio, TX) was lightly packed into the wound bed and covered with a dry sterile dressing. This dressing was then changed three times a week by a visiting nurse association. The wound measurements prior to discharge showed significant improvement in comparison to the wound measurement at presentation, with the left axilla wound measuring 1.5 x 10.5 x 0.3 cm and the left chest wound measuring 3.5 x 9 x 5 cm. |
A 38-year-old male presented to the emergency room unable to bear weight on his right knee after a fall while practicing recreational sport, specifically kitesurfing. Physical examination revealed a painful and swollen knee, with joint effusion and medial ecchymosis. He had a valgus laxity at both 0 and 30 degrees, a negative anterior drawer test, a positive posterior drawer test, and doubtful Lachman test.\nAnteroposterior and lateral X-rays of the knee were performed in the emergency room, showing an avulsion fracture on the medial femoral condyle (\n). This finding, combined with the clinical presentation, pointed toward an avulsion of the MCL of the knee.\nThe patient was discharged to our outpatients' clinics with protected weight-bearing using a brace and crutches. His magnetic resonance imaging (MRI) findings were as follows: avulsion of the MCL's origin in the medial femoral condyle with the bone fragment rotated and retracted together with the MCL, trabecular fractures adjacent to the posterolateral tibial spine with a minimum cortical collapse and a severe bone edema, altered imaging of the anterior cruciate ligament (ACL) keeping a normal route, which could be explained by a partial tear of the ligament, and complete rupture of the PCL with its proximal part retracted (\nand\n).\nThe patient was offered and consented for a navigated arthroscopic reconstruction of both ligaments.\nThe PCL was reconstructed using the double-bundle Achilles allograft technique, with an accessory posterolateral portal.\nWithin the same surgery, a medial femoral incision on the internal femoral condyle right on top of the anatomical insertion point of the MCL was performed observing the avulsion of the bone fragment rotated and distally retracted together with the MCL. A release and reinsertion with bone anchors and Spike Washer was performed.\nImmediate physical therapy was prescribed, and passive range of motion was initiated. Progressive active therapy and weight-bearing were then initiated after 2 weeks.\nAt the last follow-up, 2 years after surgery, the patient enjoyed a 0/140-degree range of motion for flexion/extension (\nand\n). He had resumed his sports activity and was pain-free. At physical examination, no ligamentous instability could be detected. A new MRI was performed showing repair signs of the MCL without intraligamentous disruption. |
A 34-year-old man presented with abrupt onset of intermittent exercise-induced pain in the left calf that improved with rest. He denied any trauma to the lower extremity. Past medical history was significant for 14 years of tobacco abuse, and 1-year use of electronic cigarette. He denied history or symptomatology concerning for vasculitis, autoimmune, connective tissue, thrombotic or rheumatologic disorders.\nPhysical examination revealed normal vital signs with palpable left femoral but absent popliteal, posterior tibial, and dorsalis pedis pulses. In spite of the absence of pulses on the left lower limb, the patient’s foot did not have other findings of critical ischemia. The remainder of the physical exam was unremarkable.\nArterial duplex ultrasound showed a complete occlusion of the left popliteal artery. The patient was started on anticoagulation with enoxaparin to bridge to warfarin. Magnetic resonance angiogram of abdomen and pelvis with three-vessel runoff revealed widely patent common femoral, profunda femoral, and proximal-mid superficial femoral arteries. However, distal segment of the left superficial femoral and above-knee popliteal arteries showed abrupt occlusion, with the occluded segment spanning 9 cm (). Mild medical deviation of bilateral above-knee popliteal artery was also present suggesting extrinsic compression laterally, although with patent right arterial system (). This deviation was concluded to be related to the proximal and lateral aberrant insertion of the medial head of the gastrocnemius muscle in the left leg and due to a fibrous band or muscle slip in the right leg (–). Catheter-based angiogram was performed 3 days later and confirmed the diagnosis of distal superficial femoral and popliteal artery occlusion with possible popliteal entrapment syndrome as the etiology (). It also revealed significant infrapopliteal artery disease suggestive of thromboembolic event versus Buerger’s disease ().\nManagement was started with enoxaparin (80 mg) subcutaneous injection twice daily, aspirin (81 mg) once daily and Lipitor (40 mg) at night. Due to the severity of his symptoms, the patient underwent catheter-based lytic therapy using tissue plasminogen activator at 1 mg/h delivered through a multi-sidehole infusion catheter with heparin at 500 units/h through the sidearm of the sheath over 48 hours (), followed by left lower extremity superficial femoral artery-to-below-knee popliteal artery bypass using ipsilateral reverse saphenous vein graft tunneled beneath the sartorius and below-knee popliteal artery endarterectomy, with good results. Following surgery, the patient was continued on warfarin to complete at least 3 months of anticoagulation. A repeat duplex arterial ultrasound was performed 2 weeks post-surgery and showed a widely patent distal superficial femoral artery to below-knee popliteal vein bypass graft without evidence of stenosis. |
A 48-year-old male was admitted to the internal medicine ward with chronic pancytopenia secondary to megaloblastic anemia. He had a past medical history of hypertension, dyslipidemia, and a history of smoking.\nThree days after admission, septic bursitis of the right olecranon was diagnosed and the patient was treated by surgical drainage of the abscess and a week-long course of antibiotic therapy (fluoroquinolone and penicillin groups). During the next 72 hours, a high fever and abdominal distention were noted. Free air in the abdomen was shown on urgently performed CT and the patient was transferred to the operating room.\nExploration of the abdomen revealed an ischemic area in the cecum with a perforation. Right hemicolectomy with ileostomy was performed. The remaining large intestine was brought out to the abdominal wall as a mucous fistula at the left upper quadrant.\nDuring the next four days in the intensive care unit, the patient continued to be septic with highly elevated temperatures (39.2°C) and extensive leukocytosis (37000 cells/uL). During this time, intensive supportive management included the administration of vasopressors and broad spectrum antibiotics.\nOn a detailed workup of sepsis, stool sample cultures for Clostridium difficile toxins from the rectum and end ileostomy were performed. Diagnosis of colitis was made by a positive Clostridium difficile toxin in the rectal sample and was confirmed by CT findings of a thickened, dilated, and distended colonic wall (mucus fistula segment) (). Clostridium difficile culture from the ileostomy was negative. Moreover, a pathological examination disclosed cecal perforation, acute peritonitis, and a tubulovillous adenoma in the cecum which was located 3 centimeters distal to the point of perforation. No evidence of colitis was encountered in the removed segment of the right colon.\nInfection disease was consulted on how to treat the colonic descending loop. A novel intraluminal antibiotic irrigation through a Foley catheter inserted into the isolated mucus fistula was implemented. The patient recovered after three weeks of intraluminal vancomycin (250 mg diluted in 150 mL of normal saline x Q6) and metronidazole (500 mg x Q8). Both antibiotics were given into the mucus fistula over 30 min. The patient was discharged from the unit four weeks after admission. |
A four-year-old girl presented to the emergency service with painful left hip and fever. There was no previous relevant medical history. There were no other local or systemic symptoms, except for a cervical adenopathy. On physical examination, she walked with a limp, and movements of the left hip were painful (mainly external rotation), but not restricted. Blood exam revealed anemia (Hb 8.7 gr/dL), normal WBC, ESB of 123 mm, and reactive C protein of 149.7 mg/L. An initial X-ray to the pelvis revealed no changes. An ultrasound of the left hip was performed revealing small infusion and synovitis. Guided puncture was then performed being macroscopically compatible with reactive arthritis, and general and bacteriological tests were demanded. Because of the unusual characteristics of the pain, a CT scan to the abdomen and pelvis was performed revealing a left adrenal mass and retroperitoneal adenopathies in the celiac trunk and superior mesenteric artery ().\nDespite the painful complaints of the patient, no bone or articular involvement was found in the CT scan. No further alterations were reported in the thoracic CT scan or in peripheral blood smears. Bacteriological examination of the hip effusion was negative. MRI was also performed. The direct myelogram was compatible with infiltration from neuroblastoma. Bone marrow biopsy and cervical adenopathy specimens were collected to perform histological diagnosis. Skeletal scintigraphy demonstrated numerous points of osteoblastic activity compatible with metastatic activity, and the 12 iodine-123 metaiodobenzylguanidine scintigraphy concluded the following: “Abdominal mass with low expression of noradrenergic transporters. Diffuse bone metastasization with high expression of noradrenergic transporters. No other soft tissue involvement was detected.” In the histological report of the cervical adenopathy, the diagnosis of neuroblastoma NOS was performed. Immunohistochemistry revealed extensive expression for synaptophysin and CD56 (NCAM) and absence of expression of myogenin. ().\nBone marrow biopsy revealed extensive metastatic involvement. The patient started chemotherapy two weeks after admission, with 8 cycles of rapid COJEC protocol. After six months of follow-up, the primary tumor was still without criteria for resection, despite a decrease in the metastatic involvement. Given the chemotherapy-related renal toxicity, it was decided to proceed with irinotecan in combination with temozolomide (TEMIRI). After thirteen months of follow-up, no significant regression of the primary tumor occurred, so surgery was contraindicated and the patient was proposed for stem cell treatment. |
She is a 17-year-old Chinese girl, the second of three siblings, who presented to the child psychiatry clinic at the age of 13 years, with prominent features of OCS. The symptoms started at the age of 11 years, during which she was obsessed with her body's physiological activities such as breathing and swallowing. In relation to that, she had compulsion to count her respiratory rate and how many times she had swallowed her saliva. She experienced intense feeling of distress when she resisted the repeated thoughts and behaviour. This occurred after her grandmother passed away and a year before her sixth-grade examination which was considered important in the Malaysian education system. However, the symptoms did not affect her social and academic functioning then. There was no history of psychiatric illness in her family.\nTwo years later, at the age of 13, the symptoms worsened. The obsessional theme changed from counting her breaths to thoughts of death and compulsion of cursing the dead, triggered by seeing the graveyards. As a consequence, she developed significant symptoms of depression including suicidal ideas. Later, she developed delusion of reference. Her social and academic functioning deteriorated and she was admitted to psychiatric ward for a month period. A diagnosis of severe OCD with comorbid major depressive disorder (MDD) with psychosis was made. She was treated with escitalopram 10 mg nocte and risperidone 0.5 mg bid. Sand tray therapy and cognitive behavioural therapy (CBT) were commenced later. The symptoms of OCD and MDD improved and she was discharged after a month of admission.\nDespite good compliance to treatment, she never attained full remission. Subsequently, she developed psychotic symptoms characterized by delusions and disorganized thoughts and behaviour. It started with repeated doubts of the existence of God, ghost, and the holy spirits and the compulsion to get reassurance over her doubts. Later, the thoughts became delusional in nature, when she started to believe that God, ghost, and the holy spirits were watching over her all the time. She felt frightened and because of these beliefs she could not bathe on her own. She believed there was a CCTV placed in her house to spy on her. She could not go out from her home as she believed people were looking and talking about her. She never had mood or negative symptoms.\nThe diagnosis was revised to comorbid schizophrenia and OCD. The antipsychotic was changed from risperidone 0.5 mg bid to aripiprazole 5 mg daily in view of better prolactin profile and the fact that second-generation antipsychotic (SGA) like risperidone was understood to aggravate OCS in schizophrenia. Aripiprazole was gradually increased by 5 mg in two weeks up to 30 mg daily. However her delusion still persisted. Haloperidol 5 mg was then combined with aripiprazole for an adequate duration but the psychosis and OCS were still the same.\nSince aripiprazole was already optimized, the next step taken was to substitute aripiprazole with quetiapine 50 mg while maintaining haloperidol at 5 mg. Quetiapine was increased to 100 mg a week later and then 200 mg subsequently. However she could not tolerate the medications due to extrapyramidal side-effect (EPSE), that is, tongue stiffness. Quetiapine and haloperidol were stopped and she was given trihexyphenidyl 2 mg bid for EPSE.\nThe next drug in line was olanzapine. There was an improvement in psychosis but it was not sustained for more than a month. Later the OCS worsened relative to psychosis despite adequate dose of 20 mg daily. Due to this, olanzapine was cross-tapered with paliperidone 3 mg. It was then gradually increased to 12 mg daily over the period of 4 months. Despite this trial, the result was still unsatisfactory. Thus, electroconvulsive therapy (ECT) was commenced while maintaining the paliperidone. However there was no improvement seen even after 12 ECT. Lexapro was increased to 30 mg daily over the period of two months. There was no full remission of OCS; however it managed to put her under control.\nAlthough she had good adherence to medications plus adequate dosage and duration of treatment given, the psychotic symptoms appeared resistant as evidenced by the treatment failure of several antipsychotics. Depot injection of antipsychotic was also considered as an option but it was refused by patient and parents. Decision was then made to start clozapine. It was started at 12.5 mg daily and gradually increased up to 300 mg daily over the period of 2 months. In addition, she also developed tachycardia with a heartbeat ranging from 120 to 130 bpm, hypersalivation, and significant weight gain around 4 kg in 3 months without much improvement in her psychotic symptoms.\nThe team had finally decided to retry risperidone in view of limited options left for her. Furthermore, the dose of risperidone was not even optimized, only 0.5 mg bid during the first time. Risperidone was reintroduced at a lower dose of 0.5 mg bid and slowly increased by 0.5 mg in 2 weeks' time in view of previous history of EPSE. Interestingly, she responded fairly better when she was put back on it. At 2 mg daily, there were significant improvement in the OCS and positive symptoms; however she developed amenorrhea. There was mild delusion of reference but she was able to go out from the house and carried out her basic personal needs at home. The OCS was almost negligible that she started to think and plan about her future. Both of her parents had been very supportive of her throughout the time. |
A 45-year-old female patient presented to us with pain in both the knees since 1 year. She also had difficulty in walking. Pain had gradually increased over the period of 1 year. She had no history of the previous trauma, steroid use, meniscus surgery, alcohol intake, or smoking. Physical examination revealed local tenderness over MFC and medial tibial condyle and decreased range of motion. Laboratory investigations showed no specific findings. Subchondral fracture with articular surface collapse of MFC with joint space narrowing and varus deformity was seen on X-ray in both the knees. Sclerosis was seen on X-ray in the medial tibial condyle on both the sides. X-ray of both knees revealed SPONK in Koshino’s Stage IV and Carpintero’s Stage IV (). MRI of right knee showed characteristic focal hyperintense areas surrounded by band like hypointense areas in both MFC and MTP which was suggestive of subchondral collapse with ill-defined bone marrow changes (). Computed tomography (CT) scan of both knees was done to assess the deformity. Right knee showed a defect measuring 2.51 cm in the MFC (). Left knee CT scan also showed defect in MFC (). Taking the deformity and disability into consideration, total knee replacement (TKR) was planned for both sides. Initially, left knee was operated. Large longitudinal osteochondral flap was seen intraoperatively on MFC’s extensor surface. The MTP was depressed and cartilage fibrillation was seen. After excising the flap, there was defect seen in medial condyle with sclerotic bone at base. An auto-bone graft obtained after cutting posterior condyle was used to build up the defect (). A cruciate retaining type of TKR was done (). Two weeks later, right side TKR was done. MFC of right side also had a large osteochondral fragment which was excised to leave defect with depth more than 5 mm. This defect was reconstructed with bone graft obtained from posterior cuts. MTP of right side had similar findings as left side (). Posterior stabilized type of knee replacement component was used along with femoral extender stem (). Histopathological examination was done of the excised osteochondral fragments and tibial cuts. It revealed necrotic bone, surrounded by an area of fibrovascular granulation tissue on both the femoral and tibial sides (). Patient was mobilized full weight bearing after surgery and has good functional outcome at 2 years follow-up. |
A 51-year-old male with a history of nonischemic cardiomyopathy with a left ventricular assist device was admitted for expedited heart transplant evaluation. The evaluation included an elective colonoscopy in light of a family history of colorectal cancer in his mother who died at age 61 from the disease. The patient had his first screening colonoscopy at age 45 and was diagnosed with benign polyps, which were removed, and left-sided diverticulosis. The procedure was uncomplicated and he was advised to repeat a colonoscopy in five years.\nThe patient was without GI symptoms at the time of his colonoscopy. He denied tobacco, alcohol, or illicit drug use. His medications included amiodarone, aspirin, famotidine, levothyroxine, lisinopril, metoprolol, sildenafil, and intravenous heparin as well as torsemide, acetaminophen, docusate sodium, and melatonin as needed. On examination, he had a left ventricular assist device port entering at the upper abdomen, but otherwise the abdomen was soft and nontender to palpation with normal bowel sounds and no appreciable masses or ascites.\nThe patient underwent a standard bowel preparation which included a clear liquid diet the day prior to the procedure and 20mg of Dulcolax with 4 liters of polyethylene glycol the night prior to the procedure. Monitored anesthesia care sedation was administered with propofol. The colonoscopy was performed at night without difficulty with good bowel preparation. Abdominal pressure was briefly required to maneuver around the splenic flexure. The colonoscope was advanced to the cecum with identification of the appendiceal orifice and ileocecal valve. Findings included multiple sigmoid and descending colon diverticula and two, small (<5mm) sessile polyps that were removed using cold forceps.\nThe night of the procedure the patient had no pain or nausea and ate dinner and breakfast the following morning without incident. He then developed epigastric abdominal pain in the midmorning approximately 12-14 hours after the procedure and had one episode of nonbloody, nonbilious emesis following lunch. On physical examination, he was afebrile with a blood pressure of 104/89mmHg, heart rate of 68 beats per minute, and oxygen saturation of 100% on room air. Abdominal examination was notable for mild distension and moderate tenderness to palpation in the epigastric region without guarding or rebound tenderness and decreased bowel sounds. Laboratory examination revealed an elevated lipase of 2275 U/L and amylase of 1141 U/L. Additional abnormal laboratory findings included an elevated aspartate aminotransferase of 105 U/L, alanine aminotransferase of 94 U/L, and total bilirubin of 1.4 μmol/L (normal prior to the procedure). An abdominal X-ray did not reveal an obstructive bowel gas pattern or evidence of free air. A computed tomography (CT) scan of the abdomen/pelvis revealed diffuse edematous changes of the pancreas with surrounding inflammatory stranding in the bilateral paracolic gutters, extending superiorly to the perihepatic region and inferiorly to the pelvis (). The constellation of symptoms, labs, and imaging were suggestive of an episode of acute pancreatitis.\nThe patient was treated conservatively with bowel rest, intravenous fluids, and analgesics as needed. Over the next three days his symptoms and abdominal examination improved and his diet was advanced to a regular diet. The lipase normalized to 15 U/L. |
This is a case of a caucasian 48-year-old woman, nulliparous, in the third trimester of pregnancy, who presented a nontraumatic progressive left hip pain. She had no relevant medical history, referred no usual medication (before or during pregnancy time), and no smoking or drinking habits. She recurred to the Emergency Room(ER) of São Teotónio’s Hospital - Viseu, in April 2012, with left thigh pain with a mechanical rhythm that began at the 8th month of gestation, with progressive exacerbation and no history of trauma, denied fever, or weight loss. Physical examination showed hip pain with flexion greater than 120° and with internal rotation, with no other complaints. There were no motor or sensory deficits in the limbs. The radiograph revealed an apparent area of hypotransparency of the femoral head () and the ultrasound showed presence of moderate joint effusion. She also performed blood tests with thyroid function and inflammatory markers which were normal. She was discharged and advised to initiate conservative treatment with no weight-bearing of the limb through gait with support of crutches and painkillers. She was kept under surveillance and continued her treatment as an outpatient. During surveillance a full rheumatoid and calcium metabolic profile was studied with no abnormal results. The delivery, considering the high risk of proximal femur fractures in these cases, was performed by C-section, electively, in May 2012, at 43 weeks of gestation and without complications. In the 2–3 weeks after childbirth, the patient remained symptomatic and repeated X-ray showing a small lithic zone (2 cm × 2 cm) circumscribed to the internal femoral head () and to attain further characterization of the lesion an MRI was performed (). This examination reported a hyposignal zone in T1 and hypersignal in T2 of the femoral head and metaphyseal-diaphyseal transition of the left femur with a size of 3 cm ×4 cm and still free intra-articular fluid. A dual-energy X-ray absorptiometry (DEXA) evaluation was also performed in follow-up with her General Practitioner with femoral neck and lumbar spine T values of -2.8 and -2.1, respectively. After the exclusion of more probable diagnoses, the presence of transient pregnancy-related osteoporosis was considered more likely to be present, with vigilance and conservative treatment maintained.\nIn February 2014 (20 months after childbirth), she presented a considerable improvement of the complaints, with good mobility/function of the left hip joint, with no need for walking aid, and with a second MRI revealing reduction of intra-articular free fluid and downsizing of the extensive signal change that affected almost the entire left femoral head, which had a size of 1 cm ×2 cm (). She maintained follow-up in our outpatient consultation with complete resolution of the initial clinical presentation in April 2014. |
A 62-year-old female patient was referred by her oncologist for a routine dental evaluation. The patient’s medical history revealed a cutaneous melanoma of the left dorsum diagnosed six years before she was referred to our dental service. The tumor was primarily treated by surgery and developed two local recurrences (one and three years following surgery, respectively), which were also managed by surgical resection. Disease progression was identified five years after the first treatment and confirmed by a computed tomography (CT), which revealed multiple organ involvement including the lungs, skin (subcutaneous nodules on the dorsum) and bone (osteolytic lesions in the iliac and femur bones, ribs, vertebrae, sternum and scapula).\nThe patient was undergoing a palliative treatment protocol based on dacarbazine, zoledronic acid and radiotherapy in the lumbar region (total dose of 20 Gy) and in the left supraclavicular fossa (total dose of 36 Gy) when she was referred to our dental facility. An extraoral clinical examination identified a pigmented subcutaneous nodule on the patient’s dorsum, measuring 5 cm in diameter, and scarring from the previous surgical resections.\nThe patient’s chief complaint was the loss of the dental crown of her upper left incisor. She denied oral pain or the existence of any relevant oral soft tissue lesion. Intraoral soft tissue examination revealed a nodule with an ulcerated surface and areas of telangiectasia, on the posterior left lateral border of the tongue, measuring approximately 1 cm in diameter, adjacent to a partial edentulous mandibular area (Fig. A). Based on the clinical features of the tongue lesion and on the patient’s medical background, the diagnostic hypothesis included metastatic melanoma, squamous cell carcinoma and fibrous hyperplasia.\nAn incisional biopsy was performed under local anesthesia. The histopathological analysis showed nests and solid sheets of non-pigmented atypical cells with an epithelioid phenotype infiltrating the sub-epithelial connective tissue. A high mitotic index was observed (Fig. B). Tumor cells displayed diffuse immustaining for S100 protein. Other melanocytic markers such as HMB-45 and Melan-A were negative. (Fig. C).\nAfter the diagnosis of a metastatic lesion on the tongue, the patient was referred back to the clinical oncologist who maintained her in a palliative protocol of chemotherapy with cisplatin, dacarbazine and vinblastine (CVD). Unfortunately, the patient died due to disease progression four and a half months after the diagnosis of tongue metastasis. |
A 67-year-old Japanese man presented with dysphagia. He had a medical history of exploratory laparotomy for ileus but no history of anticoagulant medication. Upper gastrointestinal (GI) endoscopy revealed type III esophageal cancer in the middle thoracic esophagus (Fig. a). An upper GI series revealed an irregular stricture in the middle thoracic esophagus. Computed tomography (CT) revealed a primary tumor and swollen cervical and mediastinal lymph nodes (Fig. b, c). Fluorodeoxyglucose-positron emission tomography (FDG-PET) also revealed a primary tumor in the esophagus (Fig. d) and swollen lymph nodes in the mediastinum with abnormal uptake.\nAs a result, he was diagnosed with middle thoracic esophageal cancer (squamous cell carcinoma) and T3N3M0 in clinical stage III according to the criteria of the Japan Esophageal Society. He was treated with combination chemotherapy of docetaxel, cisplatin, and fluorouracil as neoadjuvant therapy. Chemotherapy for 2 months resulted in shrinkage of the primary tumor of the esophagus and lymph nodes in the mediastinum (Fig. e–g). FDG-PET revealed decreased uptake in the primary tumor and lymph nodes in the mediastinum (Fig. h).\nAfter chemotherapy, he underwent subtotal esophagectomy with three-field lymphadenectomy and gastric conduit reconstruction through the retrosternal route by thoracoscopy and laparotomy. Postoperative care was performed under mechanical ventilation in the intensive care unit (ICU). On postoperative day (POD) 1, mechanical ventilation was terminated because of a good respiratory condition. However, he suffered from dyspnea and severe acidosis and chest X-ray examination revealed right pneumothorax, which was observed 5 h after extubation. Furthermore, air leakage was observed from the water seal chamber of the chest drain system. We thought that the respiratory disorder was caused by the right pneumothorax. Therefore, mechanical ventilation was started again. After that, it became clear that the cause of the pneumothorax was the air sucked from his drain insertion site. A few sutures were added at his drain insertion site. And we decided to continue his treatment under mechanical ventilation until the respiratory problems were improved.\nOn POD 2, the pneumothorax was improved. However, he had diarrhea, fever, and low blood pressure, which were treated with catecholamines. At the time, his abdomen was flat and soft. Laboratory findings revealed worsened inflammation with a decline in the white blood cell count, deviated enzyme levels, renal failure, and acidosis (Table ). All things considered, severe sepsis with multi-organ failure was suspected. Enhanced CT revealed bowel dilation with increases in the intestinal fluid, including the gastric conduit (Fig. a). There were no findings of arterial thrombus, including in the superior mesenteric artery (Fig. b). Upper GI endoscopy revealed necrotic mucosal changes in the whole gastric conduit (Fig. c). Therefore, septic shock caused by gastric conduit necrosis was diagnosed, and an emergency operation was planned. When we explored the abdominal cavity, we found not only gastric conduit necrosis but also intermittent necrotic changes in the intestinal wall from the jejunum to the rectum (Fig. ). Based on the findings, we considered a diagnosis of NOMI. The extent of excision was determined first by macroscopy. The non-enhanced lesion by indocyanine green (ICG) fluorescence in the small intestine was excised additionally (Fig. ). As a result, we performed an excision of the gastric conduit and 2 m of the small intestine, which was followed by a total colectomy (Fig. ). After resection, the length of the remnant jejunum was 1 m and 80 cm. We considered re-reconstruction after the removal of the gastric conduit. However, we decided that re-reconstruction could not be made because the surgical stress of re-reconstruction was intolerable considering his critical status. Esophagostomy, jejunostomy, and enterostomy for nutrition were performed (Fig. ).\nThe pathological findings revealed shedding of the epithelium, hemorrhagic necrosis of the mucosa, capillary dilation of the submucosa, and transmural necrobiosis in the gastric conduit, small intestine, and large intestine, which were compatible with NOMI. Almost all of the lesions had no thrombosis in the resected specimens. After the second operation, sepsis was improved. Furthermore, 0.01 μg/kg/min of prostaglandin E1 had been administered intravenously for a week, and enhanced CT findings revealed no ischemic changes in the residual small intestine after the second operation. On POD 16, the patient was discharged from the ICU. On POD 122, he was self-discharged from our hospital after a long period of nutritional support, including total parenteral nutrition and enteral nutrition.\nThe pathological diagnosis of esophageal cancer was squamous cell carcinoma and pT3N1M0 stage III. On POD 64, follow-up CT revealed the recurrence of esophageal cancer at the para-aortic lymph node. Although, we considered re-reconstruction of the esophagus, we abandoned this strategy, because his condition could not tolerate surgical stress, and recurrence of esophageal cancer was confirmed. He died of the recurrence of lung metastasis from esophageal cancer 9 months after the operation. |
A 77-year-old female patient who was 153 cm tall and weighed 58 kg was admitted to hospital with chronic diarrhea. Abdominal and pelvis computed tomography showed that she had cholangitis due to gallbladder stones. The patient had a congenital giant hemangioma of the lower lip (). She had a history of hypertension and showed elevated liver enzymes. Other preoperative laboratory results, including coagulation tests, were within the normal range.\nLaparoscopic cholecystectomy was planned. Due to the giant hemangioma of the lower lip, airway management was expected to be difficult because it seemed like a mask would not fit. The hemangioma was 12 × 5 × 5 cm (). Its surface was dry with no visible bleeding, was soft when pressed, and did not occupy the oral cavity. Manual displacement to the caudad was possible and the lower gum was well exposed. The mouth opening was two fingers wide and we classified the patient as class 2 Mallampati. Neck range of motion, jaw translocation, and thyromental distance were normal. The day before surgery, we explained the pros and cons of various airway management methods and the possible need for invasive airway access and got written informed consent. The written informed consent of publication for the use of photos and case details in this report has been obtained. Initial vital signs checked in the operation theatre showed that her blood pressure was 156/77 mmHg, heart rate was 66 beats/min, and oxygen saturation was 97%. Intraoperative monitoring devices were a radial artery catheter to monitor arterial blood pressure, an electrocardiogram, pulse oximeter, a BIS A-2000TM bispectral index monitor (Aspect Medical System, USA), and a train of four scans (IDMED, France). An HFNC Optiflow THRIVE (Fisher & Paykel Healthcare, New Zealand) with an FiO2 of 1.0 and a flow of 50 L/min was used for preoxygenation (). During 6 min of preoxygenation, we instructed the patient to breathe through her nose with her mouth closed. Arterial blood gas analysis after preoxygenation showed that her PaO2 level was 462 mmHg and PaCO2 level was 34 mmHg. To avoid bleeding due to the desiccation and friction, we covered the hemangioma with wet gauze to keep it moisturized. Difficult airway trolley, including emergency cricothyroidotomy kit and suction device were prepared. Anesthesia induction was performed using 1% propofol and succinylcholine. During airway manipulation, HFNC was maintained with an FiO2 of 1.0 and a flow of 50 L/min. An assistant applied a backward-upward-rightward-pressure maneuver to assist in visualizing the glottis while also manually displacing the hemangioma caudad. A cuffed endotracheal tube with an internal diameter of 7.0 mm was intubated with a stylet using a video laryngoscope (KoMAC Co., Ltd., Korea). While advancing the blade of the laryngoscope, we avoided it coming into contact with the hemangioma. The vocal cord was well visualized and classified as Cormack-Lehane grade 1 even though the tip of the blade did not reach the epiglottis. Intubation was successful without desaturation or damaging the hemangioma. After connecting the ventilator, the first end-tidal CO2 was 36 mmHg 118 s after propofol administration and analysis of arterial blood gas sampled right after confirmation of proper endotracheal tube position showed that the patient’s PaO2 level was 375 mmHg and PaCO2 level was 44 mmHg. Anesthesia was maintained with desflurane and remifentanil and 20 mg of rocuronium was administered at the start of surgery. At the end of the operation, HFNC was applied, all anesthetics were stopped, and muscle relaxation was reversed with sugammadex. The extubation and the recovery were uneventful. The patient was discharged at post-operation day 3. |
A 21-year-old female presented to the ED with a history of headache and diplopia after she hit her head on the edge of the table. Immediately following the accident, the patient had a transient loss of consciousness. After about two minutes, she regained consciousness but was confused and complained of headache, blurred and double vision. In addition, the family noticed abnormality in the patient’s eye and they described it as squint.\nThe headache started suddenly and it was continuous. It was in the back of the head and neck radiating to the front of the head around the eyes. The pain was dull aching in nature with a severity of seven out of ten. It was aggravated by light, but there were no relieving factors. This was associated with diplopia, which lasted for about 10 min. The patient denied any history of change in balance, difficulty in speech, weakness, numbness, deficit of memory, taste changes, seizures, irritation, vomiting, and nausea. Past medical, surgical, social, medication, and allergy history were unremarkable. Systemic review was unremarkable.\nUpon hospital arrival, her Glasgow Coma Scale (GCS) was 15/15. Neurological examination was conducted, and it showed bilateral lateral gaze palsy which was completely resolved without any intervention after 20 min since the symptoms started. The patient spent 10 min to arrive to the hospital, and 10 min in the ED before the resolution of the symptoms. No other significant findings were recorded in general physical and neurological examination. Nonenhanced CT scan of the head was done, and it showed a nondisplaced fracture over the left occipital bone (as shown in Figure ), which extends to the left petrous bone as a transverse fracture (as shown in Figure ).\nThe patient was admitted for 24-h observation unit and given 1 g IV paracetamol infusion for her headache. She was discharged home from the observation unit on oral paracetamol 500 mg every 6 h per needed, and with a follow-up appointment at the neurosurgery clinic after two weeks. After that, the patient lost to follow-up. |
A 76-year old female caucasian patient was admitted to our department, a tertiary care centre, two hours after undergoing pacemaker implantation for Sick Sinus Syndrome in a peripheral hospital.\nShe presented in poor haemodynamic condition with pericardial tamponade, requiring mechanical CPR at the entrance of the operation-theatre. Full median sternotomy was performed due to the acuteness of the situation. Opening of the pericardium resulted in hemodynamic stabilization of the patient. At inspection of the heart, no active source of bleeding was found. After insertion of a mediastinal drainage and wound closure, the patient was transferred to the intensive care unit.\nThe first post-operative day was uneventful. Lead values including voltage levels of P-wave and R-wave, impedance and threshold in pacemaker control were within the normal range (Table ).\nOn the second day after emergency surgery, an atypical position of the atrial lead was detected in routine chest X-ray (Figure ). The subsequent chest CT scan revealed malpositioning of the atrial pacemaker lead. It was exiting the superior vena cava through a perforation, running in the transverse sinus dorsal of the ascending aorta and connecting to the roof of the left atrium (Figure ).\nBased on these new findings, the patient underwent re-operation. Under general anaesthesia, in full preparation for re-sternotomy, an approach through the existing pacemaker wound was chosen. The atrial lead was withdrawn into the superior vena cava and re-positioned into the right atrial appendage. Again, the values for the lead were excellent (Table ). Due to the slightly steep insertion into the apex, the ventricle lead was also re-positioned (Figure ).\nOver the entire course of the surgery and during follow-up, the fluid quantity in the drain was controlled to exclude any bleeding; a possible pericardial effusion was ruled out by echocardiography. The patient was transferred to the intensive care unit in stable haemodynamic conditions.\nAfter an uneventful postoperative period the patient was discharged in excellent general condition. |
A 9-year-old, previously healthy female was admitted to The First Affiliated Hospital of Wenzhou Medical University (Wenzhou, China), and presented with abdominal pain and fever. Upon examination, the patient’s temperature was 38°C, the pulse was 86 beats per minute, the blood pressure was 103/53 mmHg and the respiratory rate was 20 breaths per minute. Abdominal tenderness was identified in the right upper quadrant without rebound tenderness. The remainder of the examination was normal and the results are shown in . Abdominal CT scan revealed a cystic and solid mass with a size of 11.5×8.3×6.7 cm3 and a well-defined border, which involved the right lobe of the liver and had a non-homogeneous density (). The density was marginally and heterogeneously increased in the enhanced CT scan (). The clinical impression was that this mass represented a hepatoblastoma.\nSurgery was performed to resect the tumor. A right subcostal incision was made and, following entry into the abdomen, the ligamentum teres hepatis and falciform were incised. Examination of the future liver remnant and abdominal cavity revealed no evidence of tumor metastasis. The tumor had ruptured at the diaphragmatic surface and had adhered to the diaphragm. The surgical margin distance from the tumor was at ≥1 cm. The tumor was resected following the margins, until it was completely removed. A drainage tube was inserted under the right diaphragm and exited the body through the right abdominal wall. The abdominal wall was closed using standard procedures. During surgery, the patient had 300 ml bleeding and was transfused with 1.5 units of red blood cells.\nHistological examination revealed pleomorphically shaped tumor cells with marked nuclei and clear to the eosinophilic cytoplasm (). All the primary antibodies used in the present study are shown in . Immunohistochemical staining of the tumor cells showed positive expression of caldesmon (), cluster of differentiation (CD) 68 (), Ki67 () and vimentin (), but negative expression of α-fetoprotein (AFP), creatine kinase (CK), desmin, hepatocyte, muscle specific actin (MSA), myogenic differentiation (myod) 1, myoglobin and smooth muscle actin (SMA). On the basis of these findings, the patient was diagnosed with UESL, which was confirmed by two surgical pathologists.\nThe patient was recommended for further treatment, which was refused due to socioeconomic and psychological reasons. Thus, adjuvant chemotherapy was not initiated. Following complete tumor resection, the patient was regularly examined. Abdominal CT scans and US were performed every 3 months to monitor tumor recurrence. At the 6-month follow-up, the patient was surviving free of disease; however, at the 9-month follow-up, unenhanced CT revealed a right, low-density prerenal recurrent mass with a size of ~2.7×2.4 cm2 and a well-defined border (). Enhanced CT showed a marginally and heterogeneously increased density (). The patient did not accept any further treatment. Two months later, CT scan demonstrated that the mass had enlarged to ~3.3×3.4 cm2. A right prerenal dissemination of UESL was suspected. Based on the patient’s condition, a second surgical procedure was required.\nUpon second admission, the patient was comfortable and physical examination revealed there was no abnormal condition. The laboratory results are shown in . Complete prerenal tumor resection was performed. There was tumor hemorrhage inside. Microscopic analysis showed that the tumor was evidence of undifferentiated embryonal sarcoma recurrence. However, the patient was comfortable and physical examination revealed no abnormal conditions. In addition, the laboratory results were normal. Abdominal computed tomography scan and ultrasound were performed every 3 months to monitor the tumor recurrence. At the time of writing, it has been 6 months after the second surgical procedure and there has been no appearence of abnormalities. |
The patient was a 58-year-old man, on treatment for hypertension, who was admitted to a vascular surgery service because of an incidental finding of two aneurysms of the SMA, identified during preoperative imaging exams preparatory to repair of an incisional hernia. The patient had no abdominal symptoms and on physical examination his abdomen was flaccid and painless and with a pulsating mobile mass in the epigastrium. Abdominal ultrasonography indicated a partially thrombosed saccular aneurysm in the retroperitoneal space, with no communication with the aorta. Multislice angiotomography revealed two aneurysms of the SMA, a proximal one measuring 5.9 × 5.2 × 5.0 cm and a distal one measuring 5.3 × 3.5 × 3.2 cm ( \n ). Since multiple collateral branches emerged from both aneurysm bodies, which meant the endovascular treatment would have involved a risk of damaging the intestinal blood supply, the decision was taken to perform open surgical repair. During the operation, by explorative laparotomy, access to the retroperitoneal space was achieved after performing the Cattell-Braasch maneuver, with medial displacement of the ascending colon and part of the transverse colon, exposing the infrarenal aorta and its branches. This revealed two true aneurysms of the SMA, the larger of which was around 3 cm from the arterial ostium and the smaller approximately 2 cm from the end of the first ( ). It was also possible to observe collateral branches (right colic, ileocolic, jejunal artery, and ileal arteries) projecting from the bodies of these aneurysms. A mesenteric-mesenteric, end-to-end bypass was therefore constructed, using a dacron prosthetic graft, excluding both aneurysms but preserving branches distal of the proximal aneurysm. It was decided to ligate and resect the aneurysms – sending specimens for cultures – and their lumens were opened, revealing large quantities of intraluminal thrombi ( ). Inspection of the abdominal cavity found the intestines to be viable and free from any sign of injury. During the postoperative period, the patient suffered gastrointestinal atony, but responded satisfactorily to prolonged conservative measures. Additionally, on the fifth day after the operation, a control computed tomography revealed signs of hematoma in the hepatorenal recess, managed conservatively to resolution. The patient was discharged from hospital to outpatients follow-up in good clinical condition, 18 days after the operation ( ). There was no bacterial growth in the culture of the aneurysm segment. |
A 75-year-old Japanese man was admitted to our hospital, complaining of nausea and vomiting. His previous personal and family history was noncontributory. Findings of blood examination were almost normal, including carcinoembryonic antigen and carbohydrate antigen 19-9. Physical examination revealed cool moist skin, pulse rate of 84 per minute and blood pressure of 131/76 mmHg. Endoscopy showed marked duodenal obstruction, though the mucosa of the stenotic site seemed almost intact. The biopsy specimen obtained from stenotic site revealed regenerative mucosa without malignancy. Upper gastrointestinal contrast study showed smooth stenosis of the second part of the duodenum with a dilatation of the duodenal bulb due to elevated lesion of 3 cm in diameter (Figure. ). Abdominal computed tomography (CT) showed a homogeneously enhanced mass of 2 cm in diameter with clear margin within the wall of the thickened duodenum. The tumor was low (poorly stained) on arterial phase and well enhanced on the equilibrium phase by contrast medium. Magnetic resonance imaging (MRI) revealed low-intensity mass on T1-shifted phase and high-intensity on T2-shifted phase. No abnormal findings were shown in other abdominal organs. Under the diagnosis of duodenal carcinoma arising from descending portion of the duodenum, malignant lymphoma or gastrointestinal stromal tumor of the duodenum, pylorus-preserving pancreaticoduodenectomy was performed. On postoperative histological examination, heterotopic pancreatic tissue was detected just from the submucosal layer to the proper muscular layer, which was consisted of numerous ducts without islets and acini, and thus diagnosed as Heinrich type III heterotopic pancreas. A transition to dysplastic ductal structures was well-differentiated adenocarcinoma cells proliferating diffusely in areas ranging from the submucosal layer to the subserosa, resulting in a diagnosis of adenocarcinoma arising from heterotopic pancreas in the duodenum (Figure. ). On microscopic examination, the involvement of lymph nodes No.13 was confirmed. The postoperative course of this patient was uneventful and he was discharged from the hospital on postoperative day 30. |
Twenty-one years ago, a 60-year-old man underwent radiosurgery for an AVM in the right frontal lobe, which manifested with intracerebral hemorrhage. The size of the nidus was 12 × 24 × 13 mm (nidus volume was 1.95 cc), and the AVM was irradiated by a maximum dose of 50 Gy with a marginal dose of 25 Gy. One year later, DSA follow-up showed complete obliteration of the AVM. The patient did not suffer from any clinical manifestations and therefore follow-up at our institution was terminated; he was advised to continue follow-up with his local neurologist. Twenty years later, he agreed to participate in the present study. MRI revealed cystic changes measuring 44 × 51 × 39 mm in the right frontal region, with defiguration of the frontal horn of the right lateral ventricle and slight midline shift. Signs of repeated hemorrhage with fluid–fluid level were observed (Fig. ). After intravenous contrast administration, the wall of the cyst and the previously irradiated AVM nidus clearly enhanced (Fig. ). Neurologic examination revealed only prefrontal psychological changes. The patient was informed that surgery was needed and provided consent. Two weeks later, he was admitted to our hospital following a secondary generalized epileptic seizure (negative history) with subsequent prolonged unconsciousness, for which he was intubated. CT showed cyst formation detected previously on MRI, DSA proved AVM occlusion. After extubation, four electrographic seizures originating in right prefrontal leads were found on routine 20-min EEG. Interictally, bifrontal nonspecific abnormalities with left preponderance were present. Antiepileptic drugs (phenytoin, clonazepam) were introduced. On subsequent neurologic examination, prefrontal psychological changes were again found. Urgent surgery was indicated. A small frontal craniotomy via right supraorbital incision was performed. Linear incision of the dura was followed by microsurgical resection of the MRI enhancing lesion located superficially. The lesion had a fibrotic consistency and several small vessels were seen on its surface. These vessels tightly adherent to the cyst were coagulated and cut. During the resection of the enhancing lesion, the cyst was entered and a yellowish liquid was drained. Communication with the ventricle appeared spontaneously and the ostium was dilated deliberately. Surgery was proceeded by hemostasis and dural watertight suturing. The bone flap was fixed by a titanium plate and the soft tissue was sutured in layers. Histological examination of the resected sample revealed AVM with occluded channels, channels with recanalization, and signs of neoangiogenesis (Fig. ). Surrounding gliosis was remarkable. In the vessel walls, deposition of ferric compounds was detected (Fig. ). |
A 42-year-old male reported to our outdoor department with complaints of nonhealing ulcerative lesion in right posterior maxillary alveolus region. Intraoral examination revealed a 2.5 cm × 3 cm endophytic ulcerative lesion present distal to the right maxillary second premolar tooth and extending up to the maxillary tuberosity area and medially on to the hard palate []. Cervical lymph nodes were not palpable. The clinical findings were suggestive of malignant pathology. Contrast-enhanced computed tomographic (CECT) scan revealed an ill-defined lytic permeative lesion involving the posterior maxillary alveolus with associated enhancing soft tissue in right buccal space approximately 24 mm × 25 mm × 21 mm dimensions in anteroposterior, craniocaudal and transverse directions []. The lesion was infiltrating into the posterolateral wall of the right maxillary sinus and its floor. No extension into the masticator and infratemporal region was detected. The right submandibular and upper jugular lymph nodes showed central necrosis suggestive of nodal metastasis. Incisional biopsy confirmed it as well-differentiated squamous cell carcinoma (SCC). The treatment included wide local excision with subtotal maxillectomy using modified Weber-Ferguson approach and ipsilateral supraomohyoid neck dissection. The temporalis myofascial flap was harvested through the temporal extension of incision and the flap was tunnelled under the zygomatic arch through the infra-temporal space to reach the primary defect [Figure -]. The flap was approximated with the palatal mucosa and buccal mucosa with resorbable sutures []. Skin closure was done with 4-0 prolene sutures in the facial region and 3-0 prolene in temporal region. Application of vacuum drain done in the temporal region and neck dissection region. The histopathological examination of the specimen revealed well-differentiated SCC with negative soft tissue and bone margins. Two of the 18 lymph nodes from level IIB showed metastatic deposits. No evidence of lymphovascular invasion, perineural invasion and extranodal extension reported. Postoperative course was satisfactory with normal facial nerve functions. The patient was sent for postoperative radiotherapy. Postradiation skin discoloration and oral mucositis was observed which resolved with symptomatic medications. The patient is under regular follow up for the past 6 months. No incidence of loco-regional recurrence and distant metastasis reported. The healing of flap was uneventful and had maintained good camouflage with the surrounding mucosa []. |
A previously healthy 45-year-old woman was admitted with rapidly progressive pain in her left upper limb within one week, especially obvious swollen in the forearm. The pain was characterized as a dull ingravescence ache after onset despite conservative therapy. The deformity of left upper limb had strongly affected on her daily life. There was no history of trauma. Constitutional symptoms such as fever, anorexia, or weight loss were absent. Other skeletal examinations were normal.\nOn physical examination, multiple ill-defined swellings were palpated over the left upper limb. Some ecchymotic patches were distributed overlying the swelling. Range of motion of the left wrist was limited. No axillary lymph nodes were palpable. Laboratory investigations were normal apart from a mild elevation of the alkaline phosphatase.\nRadiological examination revealed expansile cortical thickening and sclerosis were apparently presented along the humerus shaft (). On computed tomography (CT), prominent periosteal reaction with intramedullary cortical destruction was correlated with the radiographic findings. Pathologic fractures were presented at the medial aspect of left humerus (). Radiograph of forearm showed expansive cortical thickening and aggressive periosteal reaction along aspect of the ulna and radius. Extensive osteolytic destructions were involved in the elbow joint. The lytic lesion of proximal radius was surrounded by a thin rim of sclerotic bone. The intramedullary and subcortical radiolucent foci resembled “patchy osteoporosis” (). Lamellar periosteal reaction was presented at the distal radius. The left carpal bone prominently showed multiple sharp margins of lytic lesions. The scaphoid, capitate and trapezium bones were involved. Soft tissue swellings over the abnormal bone were easily visible. (). CT image demonstrated that aggressive periosteal reaction and multiple lytic lesions were presented at the proximal diaphysis of left ulna and radius. Cortical resorption and lytic destruction was obviously noted. ( & ). Because the lesion had an aggressive appearance on the plain radiographs and CT, a biopsy was performed to confirm the diagnosis. Histological examination of the specimen showed abundant intermediate-to-large ectatic vessels within a background of fibrous connective tissue intermixed with abnormal muscle fascicles. The marked proliferation of thin-walled capillaries was observed in the lesions (). The dilated vessels were embedded in connective tissue and small capillary-like vessels were dispersedly distributed in the stroma (). Pathologic examination revealed capillary hemangioma, and on the basis of the radiologic and clinical features, Gorham's disease was diagnosed. Massive resection and radiation therapy (total dose 3060 cGy) was planned. Two years after operation, there was no evidence of tumor recurrence. |
A 20-year-old female presented to our center with a telangiectatic osteosarcoma of the humerus. There was no history of malignancies within the family.\nMRI examination and open biopsy were performed by the previous physician. At the time the results were thought to be malignant lymphoma. After several diagnostic trials, the patient visited our center to seek advice about her telangiectatic osteosarcoma (A).\nWe observed a circumferential mass on the distal part of the right arm with a slight deformity of the arm, with marked venous engorgmnt and distal edema. The mass was warm and solid on palpation. Function of the right hand was still preserved. From the laboratory findings, there were marked elevation of the alkaline phosphatase and lactate dehydrogenase. From humerus X-ray, there was mixed lesions along the humerus with pathological fracture on the midshaft (A). T2-weighted MRI showed iso-hyperintense and expansile lesions along the humerus (A). For the metastatic workup, chest X-ray showed no coin lesions or metastatic characteristics. Patient also had a PET scan and the result was unremarkable. From the Clinico Pathological Conference (CPC) forum, it was concluded that the diagnosis was osteosarcoma of the right humerus stage IIB according to the Enneking classification.\nPatient took neoadjuvant chemotherapy regimen with Doxorubicin and Cisplatin for three cycles from January to March 2016. At the end of the third cycles, clinical and radiological evaluations were performed. Clinically the mass was not getting bigger compared to before chemotherapy (B). From x-ray, the mixed lesions became more marked compared to the previous x-ray (B). MRI showed that the mass was slightly became larger compared to the previous MRI with no involvement of neurovascular bundle (B).\nSix months after the initial complaint, the surgery was performed. We used extensive deltopectoral approach with anterolateral extension through the proximal part and curved backward to complete the posterior distal humerus and elbow approach. This approach was used because of the previous biopsy tract was in the posterior aspect of the distal humerus. The vascularity, rotator cuff tendons, biceps and triceps muscle, and majority of the nerves (musculocutaneous, radial, median, and ulnar nerves) were spared during the total resection of the humerus. Axillary nerve was sacrificed during the tumor resection. To reconstruct the humerus, both long shoulder hemiarthroplasty and total elbow prostheses were used. These two prostheses were joined using two long one-third tubular plate that worked as the long stem augmentation. Extension cerclage wire was used to make the implant as one unit (A). Finally, the stem was augmented with a bone cement from proximal through the distal. Prolene mesh then was sheathed to the bone cement, and then the preserved rotator cuff tendons and biceps and triceps muscle were sutured back with the Ethibond sutures (B). The resected tumor and humerus were then sent to the lab for histopathologic analysis (C).\nFrom the post-operative x-ray, the modified prosthesis sat well on the shoulder and elbow joint (). Function of the hand was excellent post-operatively. Patient also could immediately flex her elbow. Post-operative histopathological examination showed telangiectatic osteosarcoma with HUVOS IV (). After the wound healed without complication, patient underwent adjuvant chemotherapy. Two months after the surgery, patient could start writing with her right hand without marked difficulties. Further follow up of three years post-operatively, patient already came back to work and were able to perform daily activities without difficulties. Patient’s shoulder abduction and elbow flexion was shown on the pictures (). The MSTS functional score for the upper limb scored 83% which was excellent. There were no post-operative complications and the immunohistochemical workup used to rule out lymphoma (CD20, CD15, CD 30, and CK) would be planned. |
We reported a case of an 11-year-old boy referred to our center for TLE because of failure of the PM lead (Medtronic Capsure MRI SureScan 5086, active fixation). At 2 years old, he underwent surgical correction of a complete atrioventricular (AV) septal defect complicated by complete AV block. A single-chamber transvenous PM was implanted through the right subclavian vein, because of persistent left superior vena cava.\nNine years later, during a routine device interrogation, a significant increase in pacing threshold and decrease in pacing impedance of the lead were observed. Chest radiography revealed the entrapment of the lead between the right clavicle and the first rib, suggesting a possible loss of the lead insulation (Figure , Panel A). Contrast venography showed important adherences at the right subclavian and superior vena cava (Figure , Panel B). We tried to puncture the right subclavian or axillary vein, but the guidewire (including hydrophilic wire) failed to advance through the vein because of an obstruction. Due to the young age, the ipsilateral vein obstruction, the presence of persistent left superior vena cava, we decided to extract the lead and to implant a new lead.\nThe procedure was performed in a hybrid operating room under general anesthesia, arterial blood pressure monitoring, and transesophageal echocardiographic guidance. In addition, a stiff guidewire from the right femoral vein to the right internal jugular vein for potential use of the bridge occlusion balloon (Spectranetics, Colorado Springs, CO) in case of vascular lacerations was deployed. Before the procedure, contrast venography showed important adherences at the right subclavian and superior vena cava (Figure , Panel B). After the pocket was opened and the generator removed, it was immediately clear that it was impossible both to retract the active fixation screw and to advance the locking stylet to the lead tip, because of the lead fracture under the right clavicle. Thus, after prepping the lead with an extender (Bulldog, Cook Medical, Bloomington, IN, USA), an 9Fr Evolution RL, an 9Fr RL Shortie mechanical dilator sheaths (Cook Medical, USA), and an outer sheath were advanced into the right subclavian vein, but failed to advance further, because of tenacious fibrotic adherences in the superior vena cava (Figure , Panel C). After several attempts and adjustments, using first an 11Fr and then an 13Fr Evolution RL sheaths, we were finally able to advance to the vena cava, to free the lead of any scar tissue, and to withdraw it through the sheath without complications (Figure , Panel D-F). A guidewire was placed down the Evolution shortie sheath through the same vein for new PM implantation. Echocardiography showed no tricuspid valve regurgitation and no pericardial effusion. A new single-chamber PM was then implanted, from the right subclavian vein. After 3 months of follow-up, the patient was asymptomatic, the incision healed cleanly, and the PM was functioning properly. |
A 40-year-old woman with a well-established clinical diagnosis of classic FOP presented to the emergency department of a local hospital with intractable pain of the right thigh and a several week history of a spontaneous FOP flare-up of the right thigh unresponsive to prolonged prednisone therapy and narcotic analgesia.\nThe right thigh was massively swollen and tense. Passive flexion of the knee caused extreme discomfort. Distal pulses were intact. The neck, back, both shoulders, and both elbows were fused. The left hip showed slight decreased motion, especially in rotation. The left knee and ankle had a normal active range of movement. The great toes were short with hallux valgus malformations (), and the interphalangeal joints were fused.\nDoppler ultrasonography was performed for the evaluation of deep vein thrombosis and was negative. Plain films showed developmental dysplasia of both hips but no heterotopic ossification. A computed tomographic scan of the thighs revealed swelling and heterogeneity of the quadriceps musculature. The iliopsoas was enlarged (according to official radiographic reports; radiographs and scans are no longer available). Complete blood count and clotting factors were normal. The alkaline phosphatase was 147 IU/L (normal: 43–122 IU/L). The sedimentation rate was 30 mm/h (normal: 0–25 mm/h). Creatine kinase levels were not obtained.\nAn acute compartment syndrome of the anterior thigh was suspected. Intracompartmental pressures of the right thigh were measured and recorded at 110 mm of mercury in the right mid-quadriceps and 95 mm of mercury in the right distal quadriceps. The device used to measure the intracompartmental pressures was not listed in the emergency room records. An extensive fasciotomy and decompression of the anterior and posterior compartments of the thigh were performed from a single lateral incision with immediate post-operative relief of pain. A secondary closure of the skin and fascia was performed 2 days later (). The patient was sent to a rehabilitation facility for ambulatory therapy. Radiographs performed 7 months after the fasciotomy were reported as showing extensive heterotopic ossification of the lateral thigh musculature from the anterior inferior iliac spine across the hip joint down to the knee. The hip and the knee were functionally ankylosed. |
A fit and otherwise healthy male patient aged 56 years from the Asian part of Egypt, presented to the emergency department with acute upper abdominal pain with three hours duration. The patient was in agony with anxious look. Due to religious background, he did not drink alcoholic beverages. His body temperature was 37.7°C and pulse rate was 88 beats per minute. Abdominal examination revealed tenderness over the epigasrtic region and the right side of the abdomen with rigidity maximally detected all over the right abdomen.\nPlain x-ray films detected free gas under the diaphragm and abdomino-pelvic ultrasonography demonstrated free fluid in the peritoneal cavity.\nAfter the routine work up and proper resuscitation, laparotomy was performed. Widespread variable sized serosal intestinal air cysts were seen at the first look involving long segment of jejunum and ileum. All cysts were intact and were not incriminated as a cause of pneumoperitoneun or peritonitis (Figure ). Perforated duodenal ulcer, as the cause of generalized peritonitis, was repaired with direct closure and omental patch. Nothing was done for the intestinal air cysts. Metronidazol intravenous infusion was given two times daily.\nThe patient passed very smooth postoperative course and was followed up for the next eight months until the complaint of repeated vomiting was evident and food particles of eaten diets since more than two days were recognized in the vomitus.\nUpper gastro-duodenal endoscope and barium meal study were performed and revealed gastric outlet obstruction. Computed axial tomography also confirmed the diagnosis and denied any other pathology.\nLaparotomy, through the previous midline incision, was done and exploration was systematically performed and denoted hugely distended stomach with cicatrisation at the site of previous closure of perforated duodenal ulcer and the whole length of small gut was completely free from the already described pneumatosis cystoides intestinalis (Figure ). Gastro-jejunostomy with truncal vagotomy were done to bypass the outlet obstruction. |
Patient history
The patient was a 57-year-old female with recurrent breast cancer who presented to a community hospital with a chief complaint of bilateral thigh pain and weakness. She was initially diagnosed with left breast cancer at age 50. The cancer was determined to be estrogen receptor (ER) positive stage I, grade 1 breast cancer, and the patient underwent adjuvant chemotherapy [which included doxorubicin (adriamycin)] and mastectomy followed by tamoxifen for five years. This was then followed by letrozole for approximately two years. Seven years from her initial diagnosis, she developed left axillary lymphadenopathy and was given the diagnosis of biopsy-proven recurrent metastatic breast cancer ER positive 90%, progesterone receptor (PR) negative, HER2/neu negative, and she underwent a left axillary lymphadenectomy. Doxorubicin was recommended as an initial treatment course, however, the patient adamantly refused due to her concern for cardiotoxicity, even when it was offered at a lower dose. She was instead started on chemotherapy with docetaxel (taxol) and cyclophosphamide (cytoxan) every 21 days. The second cycle was given with pegfilgrastim (neulasta) to stimulate growth of white blood cells in the body and has the common side effect of ostealgia [].
Shortly after the second cycle of chemotherapy, the patient presented to the emergency center reporting significantly worse pain as compared to the first round of chemotherapy. She stated the pain began three days after chemotherapy treatment, and pain medications did not provide relief. Increased pain was noted with ambulation and palpation of thigh muscles. Her CPK was 3,607 units/L at the time of original admission for rhabdomyolysis.
Clinical presentation
The patient was referred to physical therapy (PT) after she was given a diagnosis of rhabdomyolysis, three days after admission. Upon the physical therapist’s initial encounter, the patient deferred a physical examination due to fatigue and pain. A PT evaluation was performed on the fourth day of admission to the hospital’ |
A 35-year-old woman, gravida 2, para 1, was referred to our center after the detection of a dilated CS at a second trimester ultrasonographic examination. First trimester findings were reported to be unremarkable, and NT was 1.6 mm. A thorough examination was then performed by an experienced fetal echocardiographer to assess any potential cardiac anomaly at 27 weeks of gestation. The four-chamber view revealed a normal heart with symmetrical chambers and normal atrioventricular associations except for the presence of CS dilatation (Fig. ). In addition, the bilateral outflow tract views and the three-vessel tracheal view showed no evidence of abnormalities. During transverse scanning, the echocardiographer undertook an extensive examination to determine the normal drainage of the pulmonary veins and the non-existence of a persistent left superior vena cava (LSVC). A further evaluation to determine the reasons for the dilatation of the CS was carried out by scanning around the CS. When the sound beam was turned to section through the parasagittal plane of the fetus, a slim vessel was visualized draining into the CS; this was confirmed to be an aberrant DV as it originated from the UV (Fig. ). Two additional movie files show this in more detail (See Additional files & : Video). Other major veins-including the UV, hepatic and portal veins, and the inferior vena cava-showed a normal course and drainage.\nTo clarify the diagnosis, 3D cardiac volumes were acquired with sagittal sweeps using HDFI over the upper abdominal and thoracic regions. The acquisition time was 12.5 s, and the sweep angle was set to 35°. The volumes were immediately reconstructed and displayed in a cine loop in multiplanar mode and then stored for later offline analysis using PC software (4D Viewer, version 14.0; GE Medical Systems, Zipf, Austria). Adjusting and rotating the images in the three orthogonal planes were used to facilitate a better reconstruction of the 3D image. A combination of smooth-surface and gradient-light algorithms and postprocessing adjustments was adopted to improve the quality of the 3D color-rendered image. The origin, course, and final drainage of the DV were clearly shown in the 3D image. The spatial relationships of the associated vessels were also demonstrated (Fig. ).\nBased on the two-dimensional (2D) and 3D diagnosis of an aberrant DV communicating with the CS, amniocentesis for karyotype determination was requested for the patient; the results revealed a normal karyotype. Normal fetal growth was revealed by follow-up examination at 32 weeks of gestation, and the remainder of the pregnancy and delivery was uneventful. The weight at birth was 3242 g, and the neonate appeared healthy with Apgar scores of 10/10. Postnatal echocardiography performed on day 7 showed a normal cardiac anatomy except for a dilated CS, and the DV was closed and imperceptible. No further abnormality was noted, and cardiac systolic function appeared normal. |
A 43-year-old man with a history of dilated cardiomyopathy (functional class IV) who poorly responded to full-dose treatment for heart failure was admitted to Masih-Daneshvari Hospital for heart transplantation in August 2012. The mean pulmonary artery pressure (PAP) of the patient was 24 mmHg (peak PAP = 35 mmHg), and echocardiography showed an ejection fraction of 15%.\nThe donor was a previously healthy 22-year-old man who died of traumatic brain injury. The evaluation of the donor indicated a compatible blood group with the patient (both B+) with acceptable body surface area.\nThe transplantation was performed via orthotopic bicaval heart transplantation technique. A patent foramen ovale (PFO) was found in the heart of the donor in the operating room, which was closed up. The duration of clamping ischemia was 130 minutes. The heart transplantation procedure was performed properly with no unusual complication, and the patient was transferred to the intensive care unit (ICU) and was extubated after 2 days.\nTen days after heart transplantation, the patient complained of pain in the upper left limb, ague, and sweating. Initial assessment indicated that he was febrile with tachycardia. Examination of the extremities revealed coldness with decreased sensation in the left upper limb. Paraclinical assessments showed leukocytosis (white blood cell = 35000/mm3 and neutrophil = 87.4%). Doppler ultrasound of the left upper limb was performed to rule out arterial occlusion. In Doppler, an obstruction due to clot formation in the brachial artery, 2 cm away from the axillary fold, was confirmed.\nWith regard to the urgency of the condition, the patient was prepared for embolectomy of the brachial artery. A Fogarty arterial embolectomy catheter was utilized, and a clot and some tissue-like material were removed. The normal blood flow of the brachial artery was reestablished. After a few minutes, the pulses of the brachial and radial arteries disappeared once again and new arteriotomy and embolectomy were performed at the same place. Again, the same material was removed, and normal blood flow was returned. A day after the procedure, transesophageal echocardiography (TEE) was carried out and showed multiple hypoechoic and mobile masses in the left atrium (LA) above the left atrium auricle and within the right atrium (RA) just adjacent to the inferior venae cava ostium (cardiac suture lines). Moreover, a 2 × 2 cm loculation in the posterior aortic wall in the transverse pericardial sinus was seen. All of the valves were normal. Plain brain computed tomographic (CT) scan was performed to assess the probable presence of cerebral embolism and it had no apparent abnormality. In addition, chest spiral CT was conducted to investigate the possibility of pulmonary embolism and it demonstrated no active parenchymal infiltration and no pleural effusion.\nGiven the TEE result, the patient was prepared for emergent surgery to remove the heart masses. Both venae cavae and aorta were cannulated. Within an aortic clamping time of 25 minutes, the RA was opened and the masses on the suture line and near the inferior vena cava were removed. The tricuspid valve was normal. The septum was assessed for the presence of the PFO and it was completely closed. Then the septum was opened, and the mobile masses were removed at different points in the LA. The mitral valve was also normal. All spaces in the LA and RA were irrigated with saline. The septum was closed in two layers and air was drawn. Finally, TEE was performed and no lesion was seen. During the surgery because of coldness and disappearance of the radial pulse in the left upper limb, the Fogarty catheter was used in the previous site of arteriotomy once again and a large clot was removed from the brachial, radial, and ulnar arteries.\nThe patient was transferred to the ICU with a good hemodynamic condition. Frozen samples showed an exudate fluid with a great number of white blood cells, which favored infection. Culture of the samples was positive for Acinetobacter baumannii. After 7 days of observation in hospital and proper antibiotic regimen, the patient was discharged home with no additional complaints and normal physical examination. A 3-month follow-up showed normal good condition of the patient without the recurrence of the symptoms. |
A 72-year-old man with ischemic cardiomyopathy, severe left ventricle systolic dysfunction (ejection fraction = 31%), a wide QRS duration (146 ms), NYHA class 3 (on optimal medical management), and monomorphic ventricular tachycardia with presyncope was referred to undergo an implantation of a CRTD.\nThe pocket was created subcutaneously on the left anterior chest. The shock lead was inserted into the right ventricular apex by a cut-down method of the left cephalic vein, and the right atrium lead and left ventricular lead were inserted by an extrathoracic puncture of the subclavian vein.\nAfter the operation, no hematoma or symptoms of infection were observed. However, he complained of severe pain irradiating to his left posterior shoulder, so flexion and abduction of the left shoulder were severely limited. Same as the patients implanted CIED, we considered the pain was simply associated with the skin cutting or the contracture of the shoulder joint. Re-operation to shift the generator was thought to be one of the choices; however, the early re-operation has been reported to increase the risk of the CIED infection. So, he was treated with noninvasively and encouraged to move the shoulder joint positively to prevent the worsening of contracture. However, his pain could not get better for 6 months after the CIED implantation.\nWe consulted with an anesthesiologist, and he estimated that the culprit nerve was the supraclavicular nerve (arising from the third and fourth cervical nerves; C3, C4) or intercostal nerve (arising from the first to third thoracic nerves; T1 to T3) in terms of the dermatome distribution of the anterior chest. Furthermore, he also suspected the brachial plexus (C5–C8, T1) was involved, as the CIED leads were inserted through the subclavian vein, which is near the brachial plexus. And he also suspected that the pain was caused by the direct compression of the deltoid muscle, the shoulder joint and the clavicle, which are mainly innervated by the axillary and suprascapular nerves that originate from C5 and C6.\nFirst, he underwent a pinpoint anesthetic block of the proximal C4 by an infusion of low-dose mepivacaine (2%, 1.5 mL) by a deep cervical plexus block pinpoint, and the pain vanished dramatically. Second, he underwent a local anesthetic block of the left brachial plexus by a supraclavicular approach (1% mepivacaine 10 mL), but the pain did not vanish. Third, he underwent a pinpoint anesthetic block of the proximal C5 and C6 (1% mepivacaine 1.5 mL for each), but the pain remained.\nFrom the above results, we made a diagnosis of compression of the supraclavicular nerve by the generator. When the generator was shifted toward inner side from the skin surface manually, the pain disappeared. So we considered that the supraclavicular nerve was compressed around the cephalic vein or the shoulder joint rather than the center of the pectoralis major.\nWe planned the re-operation without the use of sedation, to confirm the elimination of the pain by the shift of the generator. We re-created the pocket and shifted the generator toward the inner side, and the pain disappeared. So we judged the pain was not associated with the leads of the CIED, and we did not remove them (Figure ). After the operation, the patient became free from irradiating pain. |
A 74-year-old female patient presented with a locally advanced tumor of the proximal pancreatic body. The patient showed no signs of obstructive jaundice and did not require biliary stenting. Initially, her tumor was noted to involve the major visceral vessels, including the celiac artery, portal vein, and splenic artery and vein (). The tumor was deemed unresectable and the patient underwent extensive chemotherapy with multiple cycles of gemcitabine, Abraxane, 5-fluorouracil, Alloxantin, Avastin, and Xeloda. After showing a favorable response, she was referred for surgical re-evaluation. The post neoadjuvant chemotherapy CT scan () showed a poorly defined infiltrative pancreatic neck and body mass measuring ∼25 × 15 mm with ill-defined soft tissue encasing the proximal splenic artery, common hepatic artery (CHA), distal celiac axis, and superior mesenteric artery (SMA). The mass also partially encased the portal vein and superior mesenteric vein (SMV). There was no radiologic evidence of hepatic metastasis and there was mild pancreatic duct dilatation. Pre-treatment and post-treatment serum CA 19–9 levels were 46 and 9, respectively. The patient did not receive any additional studies to assess GDA flow or the need for preoperative coiling/embolization. The absolute need for resection of the celiac axis was not determined until the time of the operation. The patient's functional status, perioperative risk, and likelihood of response were weighed and she was determined to be a candidate for an attempt at a modified Appleby procedure. After being properly informed of her various treatment options, she elected to undergo the operation.\nExploration of the duodenum and pancreas revealed a firm mass in the body of the pancreas with a soft pancreatic neck. No gross evidence of metastatic disease was present. There was a benign appearing lesion in the superior aspect of liver segment II, which was excised in its entirety and confirmed to be benign. Cholecystectomy was performed, followed by Kocherization of the duodenum and dissection of the pancreas and major vessels. We were fastidious in preserving the GDA. The splenic artery was controlled distal to the tumor, leaving a normal CHA pulse. The spleen and pancreatic body and tail were then serially elevated out of the retroperitoneum. The aorta was exposed, taking down the diaphragmatic crura. The celiac artery was identified at its origin, tied, divided, and oversewn with 5–0 polypropylene suture. Abnormal soft tissue was palpated along the proximal CHA. In light of this vessel involvement, we divided the distal CHA near the GDA. We then dissected the specimen off the SMA successfully and divided the pancreatic neck.\nThe inferior mesenteric vein was then clamped and tied. The inflamed pancreatic neck and proximal body were then dissected free from the right lateral aspect of the SMV and portal vein. The splenic vein was taken flush with the SMV, and its stump was oversewn, leaving good forward flow of the SMV to the portal vein. The proper hepatic artery (PHA) and the GDA were preserved throughout the resection. A Doppler ultrasound probe was used to test the GDA and PHA. Both arteries had adequate signal and in fact, the PHA had a faint palpable pulse. The liver parenchyma was also found to have a strong arterial signal. The specimen had two short stitches and purple dye placed at the neck margin and the rest of the specimen was inked per our Jefferson protocol.\nPathology of the resected specimen showed ductal adenocarcinoma with marked treatment effects with invasion of tumor into the peripancreatic soft tissue. The excision margins were free of neoplasia and the specimen had no regional lymph node metastasis (0/26). The tumor was within 2.0 cm from the pancreatic resection margin and within 0.1 cm of the circumferential margin in the posterior peripancreatic tissue. The maximum diameter of the tumor was 3.0 cm in size.\nIn the initial postoperative period, the patient had a transient transaminitis. A postoperative hepatic vascular ultrasound showed good hepatic arterial flow through the PHA and the transaminases normalized. The patient was discharged on postoperative day 9. She remains well without evidence of disease 7 months postoperatively, and has elected to receive no further chemotherapy. |
The second patient is a 72-year-old woman with a past medical history including hypertension, type II diabetes, and obesity, who presented with weight loss and steatorrhea. As her symptoms were suggestive of pancreatic exocrine insufficiency, and with the associated weight loss, she underwent a CT scan for additional evaluation. This demonstrated a suspicious 2.5-cm complex cystic pancreatic head mass with associated pancreatic gland atrophy and a dilated pancreatic duct. She was also noted to have fatty infiltration of the liver (although not overt cirrhosis) and ascites.\nShe was then referred for further evaluation. Magnetic resonance imaging (MRI) and magnetic resonance cholangiopancreatography demonstrated diffuse dilation of the pancreatic duct up to 7.5 mm with an associated cystic mass in the head of the pancreas, suggestive of main duct IPMN (). EUS was attempted, but was unable to be completed due to severe tortuosity of her esophagus. Positron emission tomography (PET) demonstrated heterogeneous metabolic activity within the pancreatic uncinate process and to a lesser degree within the body and tail. Additional workup included a paracentesis to evaluate the ascites, which was unremarkable and ultimately deemed secondary to poor nutritional status from her exocrine insufficiency. Laboratory results were within normal limits with the exception of tumor markers and a mildly low albumin (3.4 gm/dL). Carcinoembryonic antigen was elevated at 6.0 ng/mL, and CA 19-9 was elevated at 46.7 U/mL. She was placed on pancrelipase, which resolved her symptoms of exocrine insufficiency and helped improve her nutrition.\nDue to the concern for main duct IPMN, she was referred for surgical resection. Again, it was unclear if the diffuse dilation of the pancreatic duct was secondary to involvement by main duct IPMN or due to proximal pancreatic duct obstruction. The patient was planned for a Whipple procedure with possible total pancreatectomy if high-grade dysplasia was noted intraoperatively at the resection margin. Upon transection of the pancreatic neck, IPMN with focal high-grade dysplasia was indeed noted at the margin. Due to the abnormal preoperative PET scan, in addition to the high-grade dysplasia at the margin, a completion pancreatectomy was performed. Due to her poor nutritional status preoperatively, a gastrojejunal feeding tube was placed at the time of the operation. She had an unremarkable postoperative course and was discharged with feeding tube supplementation.\nHer final pathology demonstrated a 1.1 cm well-differentiated mucinous colloid carcinoma arising in a background of IPMN (intestinal type) with foci of high-grade dysplasia and intermediate grade dysplasia throughout the remaining pancreatic duct (). Staining of the tumor was positive for MUC1, MUC2, and MUC5. All margins were negative. Zero of 24 lymph nodes was involved with tumor. Her final pathologic staging was pT2N0Mx. She was referred to medical oncology for discussion of adjuvant therapy and will be initiating gemcitabine adjuvant chemotherapy in the near future. |
A 7-year-old boy was admitted to our hospital in April 2019 due to whitening of the pupil area in the right eye after a fall, which was accompanied by eye pain, headache, and vomiting for 3 days. Best corrected visual acuity (BCVA) was 1.0 in the left eye. However, only the perception of light was possible in the right eye. The right eye also exhibited mixed conjunctival hyperemia (+++), mild corneal edema, an absence of wrinkles in the endothelium, a shallow anterior chamber, an irregular pupil shape (diameter: 2 mm), an absence of the light reflex, a rough lens surface, and pigment adhesion (Fig. ). Intraocular pressure in the right eye was 42.3 mmHg, while that in the left eye was normal. Upon admission to our hospital, he was given a preliminary diagnosis of traumatic cataract with secondary glaucoma of the right eye. However, during his hospitalization, we learned that he had been diagnosed with congenital cataract of the right eye at a local hospital 7 years earlier. Therefore, the diagnosis was revised to glaucoma secondary to congenital cataract of the right eye. Treatment with intraocular pressure-lowering drugs proved effective. However, B-mode ultrasound examination revealed a wide echogenic band extending from the back of the lens to the front of the optic disc in the right vitreous cavity (Fig. ). Axis length for the right eye was 26.32 mm, while that for the left eye was 22.30 mm. Ultrasound biomicroscopy (UBM) revealed an anterior chamber depth of 1.05 mm in the right eye. UBM also indicated upward bulging of the right iris at 3–7 points. The angle had closed, the posterior chamber was no longer visible, and the anterior capsule echo of the lens was irregular. Analysis of flash visual-evoked potentials (F-VEP) revealed delayed P2 and P3 peaks in the right eye, relative to those in the left eye. Given that B-mode ultrasonography also revealed fibrous vascular membranes around the lens, the diagnosis was again revised to congenital cataract, PHPV, and secondary glaucoma of the right eye.\nSurgery was indicated due to the presence of acute glaucoma and abnormal lens morphology. Cataract extraction and anterior vitrectomy were thus performed under general anesthesia. Prior to the operation, the diameter of the dilated pupil was 5 mm, and the pupil was irregular in shape. In addition, we observed posterior synechia of the iris and massive vascular tissue integration within the lens (Fig. ). During surgery, after detaching the posterior synechia of the iris, we noticed deficiencies in the anterior capsule of the lens. The lens appeared wrinkled and had a diameter of approximately 3 mm (Fig. ). Substantial integration of vascular tissue was observed in the posterior capsule of the cataractous lens. Following posterior capsulotomy, the red light reflex disappeared. We then performed anterior vitrectomy.\nPathological examination revealed that the right lens and capsule contained little connective tissue. We also observed fibrous tissue hyperplasia, hyaline degeneration, calcification, and follicular cystic changes, in addition to active proliferation of fibrocytes (Fig. ).\nBCVA remained unchanged in either eye following surgery. Postoperative examination further revealed a transparent right cornea. The right anterior chamber was deep, the pupil was round, pupil diameter was 3 mm, the lens was absent, and mild vitreous opacity was observed. Fundus examination revealed blurring of the boundary with the right optic disc. The cup-to-disk (C/D) ratio was approximately 0.1, and the retina exhibited applanation. The blood vessels were well-shaped, and the macular structure was generally normal (Fig. ). Optic OCT indicated that the thickness of the retinal nerve fiber layer (RNFL) was 0 μm, although the macular structure of the right eye was normal. Intraocular pressure was 17.3 mmHg in the right eye.\nNeither intraoperative nor postoperative examinations revealed characteristics of PHPV such as connection of the vitreous fibrous vascular membrane to the optic disc, shrinkage of the eyeball, or retinal dysplasia. Furthermore, the lens and capsule were severely fibrotic, with an abundance of surrounding vascular tissue. The final diagnosis was anterior PHPV with secondary glaucoma in the right eye. Although further visual rehabilitation training was recommended, his parents declined. At the 6-month follow-up, visual acuity in the right eye had not improved, and findings in the remaining anterior and posterior segments were identical to those of the postoperative examination. |
A 66-year-old male (height 5'2”, weight 50.9 kg) with a history of severe idiopathic pulmonary fibrosis with two spontaneous pneumothoraces and a persistent bronchopleural fistula underwent a successful leak repair via pleurodesis. Following the procedure, the patient developed acute respiratory failure with persistent hypoxia requiring intubation and mechanical ventilation. An echocardiogram showed right ventricular strain with an EF of 78%, right ventricular systolic pressure of 115 mmHg, and enlarged pulmonary arteries, suggestive of obstructive shock from a pulmonary embolism or pulmonary hypertension. Limited improvement prompted the need for monitoring beyond that provided with transesophageal echocardiogram. Initiation of a right heart catheterization with a pulmonary artery catheter allowed continuous real-time monitoring of hemodynamics and assessment of pulmonary capillary wedge pressure, to further guide therapy towards a potential lung transplant.\nA critical care fellow, under the supervision of an attending physician, used an ultrasound-guided technique to successfully place a 9 Fr introducer sheath [Arrow International, PA, USA] in the right internal jugular vein, through which the pulmonary artery catheter [Edwards Lifesciences, CA, USA] was inserted. During the initial attempt to float the catheter, right atrial, and ventricular wave forms were confirmed with the catheter advanced 20-30 cm, and a pulmonary artery wave form was confirmed at about 40-50 cm. Pulmonary artery pressures were measured varying between 100/30 mmHg, but wedging of the catheter was unsuccessful. Subsequent attempts confirmed appropriate right atrial and ventricular wave forms but failed to advance through the pulmonary artery. During the final attempt, the catheter was advanced to about 40-50 cm without acquiring a pulmonary artery wave form, but upon attempted removal, it stopped at approximately 30 cm, without any possible further retraction. The inability to remove the catheter prompted the use of imaging to evaluate catheter misplacement due to possible congenital anomalous anatomy or intravascular entrapment.\nPortable chest x-ray showed the catheter with complete retrograde looping directed upwards off the film (). A further CT Venogram of the head showed the tip of the catheter terminating within the right internal jugular vein at the level of the jugular foramen (). At this point, it was thought that the catheter had become entrapped via looping or knotting within the vasculature of the skull, and the neurosurgical team was consulted that determined that removal would likely require a surgical procedure involving incision of the jugular vein.\nDuring the preoperative evaluation of the patient, however, it was determined that the catheter might be stuck at the tip of the introducer sheath, and it might be possible to remove the catheter first by carefully withdrawing the introducer sheath. The catheter was then removed under aseptic conditions by first withdrawing the introducer sheath over the pulmonary artery catheter. The catheter was then gently pulled in the cephalad direction followed by the caudal direction. All through the procedure, the balloon remained completely deflated and utmost caution was exercised to apply only the minimal traction necessary. Given this eventual solution, it is likely that in the initial attempt to retract the catheter, the proximal part of the loop was the first retrieved, diminishing the size of the loop with limited movement of the tip. This caused the formation of a tight kink at the exit point of the introducer sheath, thus entrapping the catheter. |
We describe a case of a 36-year-old black African woman with two previous live births by cesarean section and two previous miscarriages who was referred in her fifth pregnancy after 6 weeks of amenorrhea. Her serum quantitative β-human chorionic gonadotropin (bHCG) was 16,124 mIU/ml. However, an intrauterine or extrauterine pregnancy could not be located on a transabdominal ultrasound scan.\nA copper intrauterine contraceptive device had been removed 2 months prior to her last menstrual period. She reported using one cycle of clomiphene 50 mg with the hope of achieving a twin pregnancy.\nShe had delivered twice by cesarean section for failure to progress. Her last two pregnancies had been first-trimester miscarriages; one was managed expectantly, and the other was surgically evacuated, though the actual procedure was unknown to the patient. She did not have any chronic medical illness and was not receiving any medication prior to this presentation. She stayed in a city suburb that was well serviced. She was a school principal in her second marriage with no children in the current relationship. She did not smoke or drink alcohol.\nOn examination, she had a normal blood pressure of 113/70 mmHg and a pulse rate of 98 beats/min. Her body temperature was 37.5 °C. On examination, her cardiorespiratory and neurological systems were normal. Her abdomen was soft and not tender. The result of her pelvic examination was normal. TVUS showed a gestational sac of 13 mm with irregular margins and a visible yolk sac located on the anterior isthmic portion of the uterus, raising suspicion of a cesarean section scar ectopic pregnancy. She declined a Doppler ultrasound evaluation scheduled for the next day. She was scheduled to have serial bHCG evaluations every 48 h.\nA repeat serum quantitative bHCG done 48 h after the initial test revealed a level of 21,521 mIU/ml, a 33% rise. She defaulted follow-up until 1 week later, when she presented with pelvic pain of increasing intensity for 5 days. An urgent transvaginal scan was performed. A fetal pole with active cardiac activity (crown-rump length 0.9 cm) in a gestational sac was located in the anterior low myometrium. The sac traversed the full width of the anterior myometrium, with the posterior margin of the sac abutting the anterior margin of endometrium and the anterior margin of the sac extending to a subserosal location in a fairly exophytic fashion. There was evidence of trophoblastic circulation on Doppler examination. There was no endometrial fluid or free pelvic fluid (Fig. ).\nShe was immediately admitted for a diagnostic laparoscopy/hysteroscopy and possible excision of the scar pregnancy if confirmed. A preoperative complete blood count showed hemoglobin 13.4 g/dl, white blood cells 7.2 × 103/μl, and platelet count 243 × 103/μl. The patient’s kidney function was normal with sodium 135 mmol/L, potassium 4.9 mmol/L, urea 3.5 mmol/L, and creatinine 65 μmol/L. The patient’s liver function test results were also normal. She had a negative result in a blood test for human immunodeficiency virus. Urinalysis did not show abnormalities. The patient’s random blood sugar was 5.6 mmol at admission. At laparoscopy, the bladder was adherent high on the anterior uterine wall, and the ectopic pregnancy was not visualized (Fig. ). At hysteroscopy, there were extensive adhesions within the lower endometrial cavity, which obscured visibility. There was no active intracavitary bleeding ruling out a threatened or inevitable miscarriage. We could not visualize any obvious bulge in the cervical canal suggestive of a cervical ectopic pregnancy. Because of the uncertainty of the location of the pregnancy due to adhesions, excision was postponed. Postoperatively, the patient became unstable with low blood pressure, systolic pressure range of 82 to 95 mmHg and diastolic pressure range of 40 to 55 mmHg, and a pulse rate range of 64 to 73 beats/min, but without active vaginal bleeding or use of medications inducing persistent hypotension. Anesthesia had been induced with etomidate 16 mg and suxamethonium 100 mg, and maintenance was initiated with isoflurane 0.8–1.5%. Intra- and perioperative analgesia was induced with fentanyl 200 mg intravenously (IV), indomethacin 100 mg rectally, and paracetamol 1 g IV. Antibiotic prophylaxis was with ceftriaxone 1 g IV and metronidazole 500 mg IV. This prompted us to order an urgent MRI scan to map the location of the pregnancy in the immediate postoperative period. MRI confirmed the TVUS findings of a cesarean section ectopic scar extending to the serosa (Fig. ).\nAn emergency laparotomy was then performed on the same day. The abdomen was entered through a Pfannenstiel incision along the old skin scar. A transverse incision was made in the upper uterine segment just above the adherent bladder. The products of conception were removed with forceps, and the gap in the anterior myometrium at the old scar was seen and felt. There was massive bleeding from the implantation site. Twenty milliliters of vasopressin (20 U diluted in 100 ml) in normal saline was administered into the bleeding myometrium edges. The edges were apposed in layers with VICRYL suture (Ethicon, Somerville, NJ, USA) to repair the defect. Estimated blood loss was 2000 ml. The patient was transfused with 1 U of packed cells intraoperatively. She was continued on the same intravenous antibiotics and analgesia that had been commenced after the laparoscopy. Her hemoglobin count on day 1 postoperatively was 8 g/dl, and she declined any further transfusion. Oral iron and folic acid supplementation was commenced.\nThe patient’s postoperative recovery was uneventful, and she was discharged on day 4 after surgery. Histology confirmed the presence of decidua and chorionic villi. The patient wanted a child because she was in a new relationship, but she was no longer sure of her future fertility plans after the ectopic pregnancy. A levonorgestrel implant was inserted 2 weeks postoperatively. The patient last attended physical review at 6 weeks, and she was well with no problems related to the surgery at a telephone review at 3 months postoperatively. |
A 39-year-old lady presented with progressive continuous lower abdominal pain, abdominal distention, and low-grade fever for three days. She had four previous cesarean sections; the last one was four years ago. She was not known to have any chronic disease or allergy history. On admission, the patient presented to the emergency department in a state of low blood pressure and received immediate medical treatment. Laboratory workup showed β-human chorionic gonadotropin (β-hCG) level at 110.766 mIU/mL. Ultrasonography, done at the time of admission, revealed right adnexal heterogeneous complex mass (7 x 5 x 5 cm) and an adjacent right ovarian cyst (8 x 7 x 5 cm) with free fluid detected in the abdomen and pelvis. There was no intrauterine gestational sac. The patient underwent an immediate computerized tomography (CT scan) to assess the extent of the disease which showed large right adnexal mass with abdominal ascites and numerous peritoneal lesions (). Once the patient stabilized hemodynamically, she underwent an exploratory laparotomy. The patient consented for definitive treatment if an unexpected underlying malignancy was detected. Intraoperatively, 4.8 liters of blood was suctioned out from the peritoneal cavity. On the right abdominopelvic side, there was clinical evidence of ruptured ectopic tubal pregnancy as well as an adjacent right ovarian cyst with a smooth outer surface. However, there was no evidence of peritoneal carcinomatosis. The patient tolerated the procedure well and transferred to the surgical intensive care unit (ICU) in a stable condition. A right salpingo-oophorectomy was performed and was sent for histopathology for further evaluation and examination. The ovary measures 10 x 7.5 x 4 cm with a cystic lesion (2.2 cm in greatest dimension).\nThe outer surface of the cyst was smooth, and upon opening the cyst, it yielded clear to yellow fluid. The attached hemorrhagic and ruptured fallopian tube (3 x 2.7 cm) exhibits multiple small vesicles and fragments of placental-like tissue in the vicinity of the perforated areas (). Microscopic examination revealed diffuse villous dilatation with substantial hydropic changes. Circumferential trophoblastic proliferation with cistern formation was readily appreciated (). Besides, cytological atypia, mitosis, apoptotic debris, and vascular invasion were identified (). The morphological features were classic for complete hydatidiform mole, interestingly enough, arising in the background of ectopic (tubal) pregnancy. Apart from the molar pregnancy, the examination of the juxtaposed ovarian cyst was that of a benign serous cystadenoma. The patient was followed up strictly with serial blood workup, mainly monitoring of β-hCG levels, which showed a rapid drop to less than one mIU/mL over the subsequent months. |
A 75-year-old right-handed man who was an established patient came to the clinic with a rapidly growing papule overlying his left zygoma. The nodule looked crusted with a central keratin core which had been present for six months but had been growing rapidly for four weeks before presentation to the clinic (Figure ).\nThe patient denied any pain or trauma at the location. He had a past medical history of squamous cell carcinoma (SCC) in situ on his nose that had been treated with liquid nitrogen. He also had a history of invasive SCC of left temple that was treated by shave excision without indications of margins. There was no evidence of recurrence of SCC since it had been treated. The patient had a previous history of giant cell tumor of the distal phalanx of the left thumb two years prior to this presentation which was treated by disarticulation at the proximal phalanx of the involved digit.\nOtherwise, the past medical history, and social and family history were not significant. On review of systems, it was established that the patient had no fever, chills, cough, or any oral lesions. His medications included aspirin, pravastatin, zolpidem, calcium carbonate, and folic acid. On skin examination, the scalp and forehead had ill-defined erythematous macules with overlying adherent scales. The remainder of the physical examination was unremarkable. The condition was assessed to be a neoplasm of uncertain behavior and was suspected to be a keratoacanthoma type SCC. A shave biopsy was performed. During the same visit, five lesions consistent with actinic keratosis were destroyed using liquid nitrogen cryotherapy.\nThe final diagnosis of lesion overlying the left zygoma by shave biopsy indicated SC. The patient was informed about the diagnosis and was referred to Dermatology-Oncology.\nMohs micrographic surgery of the SC was performed, and the residual tumor was debulked. The surgical specimen showed skin with unremarkable epidermis. There was proliferation of sebaceous lobules in the dermis, which showed basaloid keratinocytes at the periphery and sebaceous differentiation in their centers (Figure ).\nThe epithelioid cells display prominent nucleoli and nuclear atypia (Figure ).\nNeoplastic cells showed mature sebaceous differentiation. Overall, architectural growth was minimally infiltrative. On immunohistochemistry, the tumor cells expressed nuclear factor XIIIa, anti-epithelial membrane antigen (EMA), and tumor protein 63 (p63), and were negative for cytokeratin 20 (CK20) and neurofilament. |
A 60-year-old man initially presented with rectal bleeding and discomfort. On physical exam, a rectal mass was initially identified as hemorrhoids. Hemorrhoidectomy was performed, and pathology showed an over 20 mm thick ulcerated mucosal melanoma extending to the margins with a high mitotic rate and the presence of lymphovascular invasion. Tumor profiling showed the malignancy to be BRAF wild-type and KIT mutated (D579 deletion). Upon referral to our institution, staging CT scans showed an enlarging anal mass, a right inguinal mass, and multiple pulmonary nodules consistent with metastatic disease. He underwent palliative trans-anal excision of the rectal mass and was urgently started on dual ICI with ipilimumab 3 mg/kg and nivolumab 1 mg/kg once every 3 weeks for a total of four planned doses. After the third cycle, he presented with a constellation of new symptoms including nausea, constipation, weight loss, fatigue, and hypotension (seated systolic BP as low as 70 mmHg systolic). ICI was held, and he was admitted for further work-up.\nHis blood pressure did not respond to an initial intravenous fluid challenge of 5 l of normal saline. There were no localizing signs of infection, leukocytosis, tachycardia, or fever, so both sepsis and cytokine release syndrome were felt to be unlikely. His examination was negative other than for orthostatic hypotension. His pupillary responses to light and accommodation, and motor and sensory examinations were normal. A cardiac workup with transthoracic echocardiogram showed preserved ejection fraction without diastolic dysfunction, no significant valvular disease, and no pericardial effusion. A cardiac MRI had no acute findings. An endocrinopathy was considered, however multiple morning cortisol levels were normal as were TSH and a comprehensive evaluation of pituitary function including LH, FSH, prolactin, and GH, thereby ruling out hypopituitarism. There was also no evidence of mineralocorticoid deficiency (normal aldosterone and renin). Other etiologies of autonomic neuropathy were investigated including a work-up for autoimmune (ANA, creatinine kinase), infectious (Lyme, syphilis, HIV), and neurologic (anti-cholinergic receptor antibodies, anti-GAD65 antibody) causes, nutritional deficiencies (B12), and paraneoplastic syndromes (Mayo Clinic paraneoplastic antibody panel), all of which were negative (Table ). MRI of the brain was negative for intracranial metastases and had no abnormalities that could explain his symptoms. There was no family history of dysautonomia, synucleopathies, or other neurologic disorders.\nThere was no evidence of volume depletion based on objective bioimpedance measures of total body water and extracellular water. On formal autonomic testing (Finapres NOVA, Finapres Medical Systems, Enschede, Netherlands), he had a low supine resting heart rate (51 bpm) and BP (91/50 mmHg). Slow deep breathing revealed blunted amplitude (4.5 bpm [normal > 7 bpm]) at a low heart rate range (45–52 bpm). Valsalva maneuver resulted in normal heart rate responses (Valsalva ratio 1.38–1.57 [normal > 1.29]) but a “flat top” blood pressure profile and absent phase 4 overshoot. This constellation of findings was indicative of significant sympathetic dysfunction and resultant parasympathetic predominance. His cold-pressor test resulted in only a modest rise in blood pressure (91/52 to 108/63 mmHg [normal: BP increase by > 20/10 mmHg]) and no change in heart rate, also indicative of poor sympathetic reserve. On orthostatic testing, systolic blood pressure dropped from supine average of 92 mmHg to 68 mmHg within 30 s of standing, and further down to 57 mmHg by the 50th second, at which time we terminated the test. His heart rate increased from 49 at baseline to 63 bpm at termination of orthostasis. Peripheral resistance averaged 860 dyn.s.cm− 5 at baseline and increased only minimally (to ~ 990 dyn.s.cm− 5) with standing. Concomitantly, stroke volume decreased from 83 ml supine to 54 ml at the end of standing (50 s), suggesting excessive venous pooling. His hypotension, chronotropic incompetence, suboptimal baroreflex-mediated responses, impaired increase in vascular resistance and significant venous pooling all indicated a loss of sympathetic tone, consistent with acute autonomic dysfunction due to an acute autonomic ganglionopathy which, in his case, was presumed to be autoimmune in nature given its development while on ICI.\nTreatment was initiated with pulse dose solumedrol 1 g IV for 6 days that was converted to oral prednisone upon hospital discharge and was slowly tapered over months. Although there was no serologic evidence of the presence of autoantibodies, seronegative cases of autoimmune autonomic ganglionopathy (AAG) have been shown to respond to intravenous immune globulin (IVIg) [, ]. Thus in addition to steroids, IVIG was administered initially as 0.4 g/kg daily for 5 days (total dose 2 g/kg), followed by 1 g/kg every 2 weeks as maintenance (Fig. ). He was also maintained on midodrine (up to 20 mg three times daily), fludrocortisone (up to 0.3 mg per day in divided doses) and sodium chloride tablets (1 g three times daily). His blood pressure gradually improved to 100/60s mmHg and several months later fludrocortisone and salt were tapered off, midodrine dose was tapered down, IVIg was discontinued, and he was maintained on prednisone 7.5 mg daily, with systolic blood pressures ranging 100–120 s mmHg and minimal orthostatic changes on variable doses of midodrine as the sole treatment agent.\nRestaging CT scans approximately 4 months after the last dose of combined ICI showed disease progression in the lung and anal region for which he underwent re-excision of the anal mass followed by palliative radiation. Nivolumab alone was restarted which was well tolerated and he did not have reoccurrence of AAG at this time. Six months later CT scans showed disease progression and he was re-induced with combination ICI, but with only 1 mg/kg of ipilimumab and 3 mg/kg of nivolumab. This led to a milder exacerbation of his orthostatic hypotension, which responded to 2 mg/kg of prednisone followed by a slow taper. After his symptoms improved, he was re-challenged with nivolumab alone but again became hypotensive and ICI was discontinued. He also developed autoimmune transaminitis that was rapidly responsive to oral prednisone. Other treatment options were offered, however he opted for hospice care and ultimately passed away from disease progression. |
This is an 86-year-old Caucasian male with a 20 year history of right-sided breast cancer. He presented to the emergency department with complaints of an ulcerated, hemorrhaging right anterior chest mass. Patient was noted to be a poor historian with a history of dementia. He was alert and oriented only to person and time.\nPertinent surgical history included a right total mastectomy 20 years ago. Patient did not receive adjuvant radiation, chemotherapy, or hormone therapy post-operatively. He also refused further medical care. Per the family, the patient developed a mass on his anterior chest wall approximately two years ago. The mass progressed in size, began to ulcerate and eventually the patient was brought to the emergency department after it began to bleed.\nThe patient’s past medical history also includes bipolar disorder, dementia, and hypertension. His surgical history is significant for a right-sided mastectomy. Current medications include: Lithium 300 mg daily. He has no known drug allergies. His mother had colon cancer, otherwise, no family history of breast cancer. Patient is married and has been retired since the age of 62. His wife suffers from new onset dementia, atrial fibrillation, chronic anemia, chronic kidney disease one son has history of bipolar disease, but is functional, and helps his parents with some of the activities of daily living. His second son lives out of state. The family has the support of a family-friend who has been helping the family through the years. Patient has a remote history of smoking cigarettes and cigars, but denies use of alcohol or recreational drugs.\nVital signs on day of admission were normal with a temperature of 97.4°F, pulse of 80, respiratory rate of 14, pulse oximetry of 100% on room air. Blood pressure was elevated at 162/88. Examination findings were remarkable for a right-sided pedunculated 8 cm × 7 cm mass with a cauliflower-like appearance. The mass was ulcerated, erythematous, malodorous, and with scant bleeding ( & ).\nLaboratory studies showed a white blood cell count 6,500, hemoglobin 12.4, hematocrit 36.2 and platelet count 178,000. Chemistry profile revealed a creatinine of 1.72 and glucose 106. The remainder was within normal limits.\nAn initial chest X-ray revealed findings suspicious for prior right-sided mastectomy, as well as a prominent superior mediastinum suggesting a nonspecific mediastinal mass or adenopathy. A CT chest showed a soft tissue mass in right chest wall measuring 5.2 × 2.75 × 5 cm with post-operative changes of the right axilla. CT Head showed no intracranial hemorrhage, mass effect or shift. CT abdomen and pelvis confirmed the anterior chest wall mass, but there was no evidence of metastatic deposits within the abdomen or pelvis. A bone scan also ruled out metastatic disease.\nAn incisional biopsy of the right breast mass was performed. Pathology returned features consistent with recurrent moderately differentiated duct carcinoma of the breast with ulceration of overlying epithelium. Nottingham combined histologic grade was T3N3M1. Breast studies returned estrogen receptor positive and progesterone receptor positive. HER-2/neu by FISH methodology was normal.\nPatient was discharged to a skilled nursing facility and started on hormone therapy with tamoxifen 20 mg daily with one course of palliative radiation. An oncology evaluation determined that patient is not a candidate for curative treatment. During an initial meeting with the family at the nursing home, the family verbalized concerns over the patient’s primary caregiver’s well being and state of health and stated she was not able to properly care for her husband. Hospice care was recommended, and family agreed. Currently the patient is in a skilled nursing facility with hospice. His wife has been admitted to the same nursing facility due to general deconditioning. |
A 46-year-old female patient presented with chief complaints of painful, sudden diminution of vision in right eye, following trauma by a wooden stick 3 days prior. On presentation, visual acuity in the affected eye was the only perception of light with an inaccurate projection of light rays whereas visual acuity in the left eye was +0.2 LogMAR. Ocular examination showed purulent conjunctival discharge with matting of eyelashes, sealed corneal perforation with corneal infiltrates, and the anterior chamber was full of the exudates []. The ultrasonography of the right eye showed dense vitreous exudates and retina was in suggesting the diagnosis of endophthalmitis. Corneal scrapings and conjunctival swabs sent for bacterial and fungal smears were negative. Diagnostic vitreous tap for bacterial and fungal smears was also done, and intravitreal antibiotics (vancomycin 1 mg in 0.1 ml and ceftazidime 2 mg in 0.1 ml) were administered. The patient was started on fortified vancomycin (50 mg/ml) eye drops 1 hourly; cefazolin (50 mg/ml) eye drops 1 hourly along with natamycin 5% eye drops 1 hourly in the right eye. The vitreous tap was also negative for bacterial and fungal smears. However, the patient developed panophthalmitis in 24 h with positive t-sign and thickened retinochoroid on ultrasonography [] that led to the painful blind eye. Considering the diagnosis of panophthalmitis and painful blind eye, the patient was counseled for evisceration. After obtaining informed written consent, evisceration was done, and the excised ocular tissue was sent for histopathological examination. Histopathological examination showed the presence of lymphocytic infiltrate with caseous necrosis suggesting granulomatous inflammation []. Staining for acid-fast bacilli was positive []. Polymerase chain reaction (PCR) of the corneal tissue also showed positivity for the tubercular antigen. The patient was investigated for any systemic focus of tubercular infection, but the results were negative. There was no past history of tubercular infection or contact. The patient was started on anti-tubercular therapy. After follow-up of 6 months, the patient was asymptomatic, and there was no systemic manifestation of tuberculosis. |
In November 2015, a 52-year-old male patient with a sternal manubrium tumor, burdened by an extended oncological history, was admitted to the Thoracic Surgery Clinic, General and Oncological Clinic, Medical University of Lodz. The patient came to the Thoracosurgical Outpatient Clinic for the first time in July 2015 complaining of sternal pains. The chest computed tomography (CT) scan revealed a right lung tumor with an extensive osteolytic change involving a soft tissue component in sternal manubrium, slightly widening beyond the back contour of the sternum. In the very same CT examination, an additional right kidney tumor was found. Positron emission tomography was performed () which revealed metabolically active changes (sternal tumor maximum standardized uptake value of fluorine 18 fluorodeoxyglucose =3.5, pulmonary tumor maximum standardized uptake value of fluorine 18 fluorodeoxyglucose =13.6). The patient denied other chronic diseases. His family history was positive as to neoplastic diseases – patient’s father suffered from gastric carcinoma, and his grandfather from chronic myeloid leukemia. The patient underwent right nephrectomy in August 2015, and on the basis of histopathological examination, he was diagnosed with clear cell kidney cancer. Due to this, changes in the lungs and sternum were considered as metastatic. In September 2015, the patient was subjected to right upper lobectomy by classical anterior-lateral approach. Final histopathological examination, though, revealed squamous epithelial carcinoma of a lung. The situation implied that the change in the sternum can be a metastasis of one of the above-mentioned neoplasms. Due to massive bone destruction and the fact of not knowing the etiology of the sternal tumor, the multidisciplinary oncological and surgical team recommended the resection of manubrium.\nOn 10 November 2015, resection of sternal manubrium was performed along with reconstruction of an individually dedicated implant made of high-molecular polyethylene. In brief, the surgical technique was as follows: The procedure was started with a longitudinal incision of the skin in the central line above the tumor. After dissection of the pectoral major muscle attachments, a tumor, of size 30×30 mm, was seen, exceeding the anterior lamina of the sternum. Costal cartilages I and II were dissected, and clavicles from sternalclavicular joints were relieved. Below the tumor, the sternum was dissected transversally maintaining the 3-cm margin of a healthy bone. The lateral side of the tumor was surrounded by unchanged osseous tissue. Moving from the bottom to the top, the manubrium together with the pathological mass was removed. The material was sent immediately to histopathological examination, which gave the diagnosis of metastasis of a clear cell carcinoma. In the place of an extensive bone defect of the upper sternal part, an earlier prepared dedicated implant of sternal manubrium made of high-molecular polyethylene was placed. The shape and size of the implant were worked out based on a special software program including the data of the patient’s CT scan. Dicom files from CT data were used for recreating the proper sternal shape in file “.stl”, in “Invasalius” program.\nUsing SolidWorks program, solid form with “parasolisd.xt” extension was achieved in “NX CAM” program which allowed for generating special codes for numerically controlled milling machine. Processing tomography with consideration to a planned resection range and after designer’s consultation with a surgeon, the final 3D implant was created (). Next, using numerically controlled milling machine, the implant was made of certified polyethylene of ultrahigh molecular mass which is rigid and nonabsorbable.\nThe patient was informed about the purpose of the study, and written informed consent was obtained to have the case details and any accompanying images published. The study protocol was approved by the Bioethical Committee of the Medical University of Lodz.\nDue to physical properties, the implant constituted a perfect base for anastomosis with bone scaffolding adjacent to sternal defect. The implant adjusted to the defect was fixed to the clavicles, sternum and ribs with the help of titanium rib plates (). The material was prepared 7 days before the operation and subjected to sterilization. The size of the implant was slightly bigger than the removed sternal manubrium and had to be trimmed during the procedure using a sharp knife and surgical file. Full stabilization of shoulder girdle and anterior chest wall was achieved. After hemostasis control, pectoral muscles were stitched with each other. The patient endured the surgery well. On the second day after the surgery, control chest X-ray was performed; no pneumothorax or fluid in the pleural cavity was found. As observed on the photograph, the plates fixing the implant to ribs did not shift. On the next day, Redon drain was removed, and on the fourth day after the operation, the patient was discharged home in a good general condition. In a final histopathological description of resected change, the diagnosis of renal cancer metastasis to the sternum was confirmed. Clavicular margins, lower sternal and costal, were free of tumor cells. The skin sutures were removed after 10 days in dispensary. During next months of follow-up, the patient denied any pains related to the sternum, and returned to his profession as a teacher (). No shoulder girdle deformity or disorder was noticed. Full range of motion in shoulder joint was conserved. Yet, in a further follow-up after 4 months, the patient was diagnosed with a next metastatic change, this time located in the left neck of the femur. The patient underwent alloplasty with inserting left hip prosthesis. Currently, 2 months after the surgery, the patient’s condition is good, and he has been under surveillance of the Thoracosurgical and Orthopedic Outpatient Clinic. |
A previously healthy 16-year-old male patient, with no history of ocular disease, presented in February 2013 after sustaining a penetrating glass injury to his left eye (OS). He described an abrupt loss of vision in the affected eye and was first transferred to a hospital where the glass foreign body was removed manually in the emergency department by a non-specialist, which possibly contributed to a further extension of his initial injury. Subsequently, he was found to have NLP vision in his OS on examination. A slit-lamp evaluation revealed a zone III injury (posterior OGI) with broad corneoscleral laceration extending posteriorly. A ruptured lens and protruding vitreous were also noted (fig. ). The patient had a distorted pupil in his OS, which was non-reactive to light (using the indirect ophthalmoscope light at the highest intensity). However, a consensual pupillary reaction was present in his right eye (OD; when light was shone on the injured globe). An initial B-scan at presentation showed a collapsed globe (fig. ). The patient was transferred to the operating room where he underwent surgical repair of the corneoscleral laceration under general anesthesia. The globe was first sutured at the limbus using nylon 10–0 sutures and a further dissection showed an extension of the laceration posteriorly toward the optic nerve. Cornea, sclera and then conjunctiva were all sutured carefully using nylon 10–0 sutures, and an eye patch was carefully placed. Oral ciprofloxacin 250 mg twice per day and prednisone 50 mg once daily were started.\nOn the first day after surgery, the eye patch was removed and an examination showed a retained globe integrity with cataract formation. The patient was started on topical Zymaxid (gatifloxacin) and Pred Forte (prednisolone acetate) every 2 h.\nThe patient was noted to have an unchanged globe integrity with rapidly increasing cataract formation when followed up on postoperative days 4, 7, 14 and 30 (fig. ). The topical antibiotic and steroids were kept at 5 times per day from day 7 until day 14 postoperatively. Then, both were tapered and eventually stopped over the following month. At 1 week postoperatively, oral antibiotics were stopped and oral steroids were tapered gradually over 1 month. A follow-up B-scan revealed a flat retina and the patient was scheduled for regular monthly follow-ups. On day 7 postoperatively, the patient was found to have an uncorrected visual acuity (UCVA) in his OS of counting fingers near face with a reactive pupil to light and a normal consensual reflex in his OD. His UCVA worsened over the following follow-up visits as the cataract formation increased.\nTwo months after the initial surgery, a repeated B-scan showed a flat retina and a clear well-formed vitreous cavity (fig. ); accordingly, the patient was scheduled for cataract extraction. The anterior capsule was partially ruptured and fibrotic secondary to the penetrating injury. Yet, circular capsulorrhexis was managed to be done with the help of VisionBlue® (0.06% trypan blue ophthalmic solution). Phacoemulsification of the cataract was performed successfully, followed by an anterior vitrectomy to remove the protruding vitreous. A posterior chamber intraocular lens was implanted in the sulcus. The procedure was well tolerated and the affected eye was unpatched on the second day. The patient was started on topical antibiotics and steroids and followed up on days 1, 3, 6, 21 (fig. ) and 6 weeks postoperatively during which the topical treatment was tapered gradually.\nThe patient reported a gradual improvement in his vision after cataract extraction. On the 3rd week after the second surgery, UCVA in his OS was 20/200 and on the 11th week, UCVA improved to 20/50 with a best corrected visual acuity reaching 20/20 with a manifest refraction: plano +1.75 × 45° (fig. ). |
A 78-year-old male patient presented in April 2015 with suspected lung cancer. Chest CT examination revealed a pulmonary space-occupying lesion in the left upper lobe of the lung (Fig. ). Following fine needle biopsy, pathology confirmed the patient had lung adenocarcinoma and EGFR (Fig. ), ALK and ROS1 were negative (Fig. ). Due to the advanced age of the patient (78) together with a diagnosis of atrial fibrillation, EGFR-TKIs was recommended as the treatment of choice. From 23 October 2015, the patient commenced gefitinib 250 mg orally once a day until the disease progressed in July 2016 when CT scan of the chest indicated metastasis in the lung. The gefitinib, everolimus combined therapy was subsequently recommended because of disease progression. However, after a few months of combined treatment, everolimus had to be withdrawn because the patient experienced severe breathlessness, cough and mouth ulcers. At that time, the patient had been taking gefitinib for 16 months until drug resistance developed on 16 February 2017 (Fig. (a)).\nThe patient had second gene mutation testing at this time which indicated EGFR T790M mutation. Osimertinib 80 mg/day orally was subsequently recommended from March 2017. After only one month of treatment, chest CT scan revealed the disease had almost completely resolved (Fig. (b)). However, severe interstitial lung disease was also confirmed (Fig. (b)). The patient was found to have severe cough and difficulty in breathing at this time and the symptoms did not resolve, even after anti-infection and anti-cough treatments were administered. The patient attended our hospital for further treatment. After reviewing his treatment history and chest xray, we considered that he was suffering from severe drug-induced interstitial lung disease and advised him to cease taking the osimertinib immediately. Unfortunately, he still continued to take the osimertinib as he considered the medicine to be effective for his tumor, and refused to tell the doctor the truth until it was found that he was suffering from further dyspnea. After anti-infection and high dosage methylprednisolone (240 mg/day) and mechanical ventilation, the patient finally died of multiple organ failure after 2 weeks treatment for severe interstitial lung disease and resultant complications. |
A 56-year-old woman diagnosed with severe PR and TR was referred to our institution. She had previously received surgical relief of congenital pulmonary valvular stenosis at the age of five years. On admission, she was suffering from shortness of breath. A systolic murmur was noted at the left sternal border in the third left intercostal space, and abdominal physical examination revealed four fingerbreadths of hepatomegaly. An electrocardiogram showed atrial fibrillation with bradycardia and low-voltage f wave. Moreover, transthoracic echocardiography demonstrated severe PR and severe TR, and significant amount of TR was owing to the tricuspid annular dilatation (41 mm) and marked right ventricle (RV) enlargement (end-diastolic dimension, 41 mm) with a TR gradient of 26.9 mmHg. The tethering height of the tricuspid valve was 12 mm (Fig. ). Additionally, the left ventricular dimensions and function were normal, the inferior vena cava was not respiratory-collapsed, and a systolic hepatic vein flow reversal was noted. Cardiac catheterization showed a mild elevation of systolic RV pressure (37 mmHg) with normal mean pulmonary artery pressure (16 mmHg). Although RV dilation (RV end-diastolic volume index 141 ml/m2) was revealed by cardiac magnetic resonance imaging (CMR), RV ejection fraction was normal (71%) and tricuspid annular plane systolic excursion (TAPSE) by echocardiography was within normal limits (28 mm), suggesting that RV function was preserved. Liver congestion was demonstrated in both abdominal echography and computed tomography. The patient was judged an acceptable candidate for surgical treatment for severe PR, severe TR, and bradycardia. We did not apply ablative surgical therapy for the atrial fibrillation because of the low-voltage f wave, which is a risk factor for surgical ablation failure.\nFollowing a sternal re-entry, a cardiopulmonary bypass was established with ascending aortic and bicaval cannulation. The right atrium and ventricle were significantly dilated. After cardioplegic arrest, the pulmonary valve was inspected via a longitudinal incision of the main pulmonary artery. The pulmonary valve was tricuspid but restrictive, which led to valvular incompetence. Furthermore, the pulmonary annulus was so small that an appropriately sized bioprosthesis could not pass through the annulus. Therefore, the incision was extended inferiorly beyond the annulus, and the right ventricular outflow tract was reconstructed using a transannular patch (Fig. ), followed by PVR with a 19-mm bioprosthesis (Inspiris Resilia, Edwards Life Sciences, Inc., Irvine, CA).\nThe tricuspid valve was inspected through a right atriotomy. The annulus was significantly enlarged. The valvular tissues were considered fundamentally inadequate for valvular competence even after the undersized annuloplasty. The tricuspid valvular tethering due to right ventricular dilatation was also recognized. We implanted a 27-mm bioprosthetic valve (Epic, St Jude Medical, Inc., St Paul, MN) leaving the leaflet tissue. The epicardial pacemaker lead was subsequently implanted, and a pulse generator was placed in the left rectus abdominis muscle. Weaning from the cardiopulmonary bypass was uneventful with ventricular pacing. The patient showed good postoperative recovery and is currently doing well one year after the surgery, without prosthetic valve dysfunction. |
An 11-year-old male Rwandan, a known patient of multiple hereditary exostoses (MHE), presented to our hospital with a 6-month history of unrelenting bone pain despite treatment with NSAIDs; he later developed (two weeks prior to admission) a dull persistent aching pain and swelling of his left lower thigh that was worse at night and unresponsive to morphine. His parents noticed that he had also lost weight and appetite. There was no history of trauma or a fall. He was diagnosed with MHE at age 3 and had been living a relatively normal life and attending school. None of his siblings or any other member of his family had MHE. His past surgical history was unremarkable. On general examination, he was well nourished, had mild pallor of the mucus membranes, no jaundice, lymphadenopathy, or skin rash. All the other parameters were normal. Examination of the musculoskeletal system revealed normal stature except for the curving deformity of the left leg. There were multiple palpable bony swellings bilaterally on the upper humeri and lower femurs. The lesion on the left distal femur was markedly enlarged and tender, with induration, reddening, and limited range of motion of his knee joint (). Repeat X-rays confirmed the presence of bony outgrowths (exostoses) on the medial and lateral aspects of the distal femurs bilaterally and left metaphyseal widening common in this condition as had been previously identified when he was diagnosed at age 3, and further investigations of the left distal femur swelling () with magnetic resonance imaging (MRI) revealed a distinct enhancing lesion in the distal aspect of the left thigh at the site of intense swelling and pain. MRI showed a lesion with hypointense signals on T1WI sequences and has heterogeneous signal intensities with moderate and heterogeneous enhancement on T1WI postcontrast study and on T2WI sequences; the lesion had heterogeneous signal intensities (Figures –). At surgery, there was a necrotic lesion, and excision biopsy at the site of the left distal femur exostosis was taken (Figures and ). Hematological evaluation was normal except for a mild leukocytosis. The results of the serum biochemical tests were also normal. We did not do genotyping for EXT-1 and EXT-2 due to lack of facilities.\nHistopathological examination revealed a characteristic fibrous cartilaginous cap with a broad base (1.293 mm thick), covering a layer of normal appearing marrow and bone below which was a tumor-forming osteoid, an osteoblastic tumor as evidenced by presence of numerous bone spicules of varying maturity. There was also marked cellular atypia, grossly pleomorphic osteoblasts in the marrow with frequent mitoses. These features were those of a high-grade osteoblastic osteosarcoma. In conclusion, histopathological revealed an osteochondroma with an underlying high-grade osteoblastic osteosarcoma involving the marrow cavity (Figures –).\nFor staging purposes, CT scans of the chest, abdomen, and pelvis were done to investigate any presence of metastatic lesions (Figures and ). There were no metastatic deposits in the lungs, abdomen, and pelvis, and this was confirmed with PET scan in India where the patient was referred for specialized bone tumor treatment including limb salvage therapy. He was reevaluated, and the diagnosis of MHE and osteoblastic osteosarcoma confirmed. Whole body PET scan showed metabolically active disease in the distal left femur 8.6 × 8.1 × 16 cms in dimension with features consistent with osteosarcoma, multiple hereditary exostoses with evidence of skip lesions, loco-regional lymph node involvement, and no distant metastases. Histopathological review confirmed the earlier diagnosis of osteochondroma with osteoblastic osteosarcoma. The conclusion was that the patient had clinically localized disease. The following treatment plan was proposed and instituted: initiation with neoadjuvant chemotherapy followed by limp salvaging surgery and finally adjuvant chemotherapy. He received doxorubicin 35.5 mgs/m2 per day (day 1 and day 2), cisplatin 60 mgs/m2 for 7 days (days 1 and 2), methotrexate 12 gms/m2 per day, etoposide 100 mgs/m2/day for 5 days, and ifosfamide 2.8 gms/m2/day for 5 days followed by Ifosfamide 3 gms/m2/day for 3 days and subsequently pegylated Interferon-α2b 0.5 mcg/kg - 1mcg/kg. He completed a total of six cycles of chemoimmunotherapy.\nOur patient is still alive and back at school with no evidence of disease after 11 months of treatment and follow-up and continues to be followed up by the oncologist. |
This is a case report of a 46-year-old female with a past medical history notable for depression, asthma, and uterine leiomyomas who presented to an urgent care with 5 days of progressive abdominal pain, bloating, nausea, and subjective fevers. The patient endorsed a several month history of gaining weight, though she attributed it to her lifestyle, accompanied with strong, intermittent, crampy right lower quadrant pain. The pain was random in onset and would dissipate very quickly. However, 5 days prior to presentation, her pain dramatically increased and was persistent in nature.\nHer surgical history included a myomectomy performed 5 years prior followed by a laparoscopic hysterectomy 2 years later. She was a regular drinker, consuming four to five alcoholic drinks per night, but stopped when her symptoms worsened and had no history of withdrawal. Family history was significant for a maternal grandmother with breast cancer, mother with skin cancer, and an uncle with colon cancer.\nUpon arrival to the emergency room, the patient was tachycardic, mildly hypotensive, and febrile to 103 °F. Her abdomen was soft, distended, and diffusely tender without peritonitis. Labs were notable for a leukocytosis of 15 K. CT of the abdomen and pelvis demonstrated moderate volume, complex fluid within the abdomen and pelvis with extensive amount of gas and peripheral rim enhancement, a thickened appendix filled with fluid, and a soft tissue/cystic lesion in the anterior abdominal wall (Fig. ).\nDifferential included pseudomyxoma peritonei with a ruptured appendiceal mucocele versus PMP secondary to an adnexal ovarian neoplastic pathology with an infectious component. Per the radiology report, the origin was unclear based upon imaging and stated a ruptured adnexal cyst should be considered given the markedly enlarged septated cystic lesions in the pelvis. The patient was resuscitated, and her blood pressure and heart rate normalized with 2 L of fluid. Given the patient’s hemodynamic stability, the decision was made to attempt conservative management with antibiotics and interventional radiology (IR) biopsy and drainage.\nOn the floor, the patient remained hemodynamically stable with intermittent fevers. Her abdominal exam also remained unchanged, with persistent pain that was under control with pain medication. It was unclear if the abdominal wall mass was neoplastic, and given our initial nonoperative approach, a biopsy was performed for tissue diagnosis to properly guide further treatment. A core needle biopsy of the abdominal wall mass demonstrated a uterine leiomyoma implant, and the cytology aspirate of the peritoneal fluid showed pools of acellular mucoid material. On hospital day 7, the patient’s leukocytosis rose to 23 K, and repeat imaging demonstrated a more organized collection with intraperitoneal air in addition to multiple thick, wall-enhancing, complex cystic multiseptated lesions. Given these findings on imaging as well as her clinical presentation, the patient was taken to the operating room (OR) jointly with surgical oncology and gynecology oncology for a peritoneal washout and bilateral salpingoopherectomy. Intraoperative findings included a large amount of mucin in the abdomen and pelvis with a pocket of purulent fluid in the mid-abdomen, a very large (12 cm) cystic left ovary, dilated right fallopian tube with enlarged right ovary, a very dilated and thickened appendix, and dense, diffuse small bowel intraabdominal adhesions (Fig. ). Her peritoneal cancer index (PCI) score was unable to be determined due to the degree of inflammatory adhesions. She underwent bilateral salpingoopherectomy given the amount of inflammation in the cecum and base of the appendix, the decision was made not to perform an appendectomy to avoid leakage at the staple line and further infectious complication. The patient’s abdomen was hostile, and no larger operation was deemed necessary at this time, as the patient was nonobstructed and the origin of PMP was not yet verified. The primary goal of the operation was to clear the infection, with the intent to return at a later date when her intraabdominal contents could be properly mobilized for a complete PCI score and an appropriate oncologic resection could be performed.\nHer hospital course was complicated by recurrent pleural effusions requiring multiple thoracocenteses, negative for cytology, and persistent need for supplemental oxygen. She also developed a secondary intraabdominal abscess requiring IR-guided drainage. After the patient clinically improved, she returned to the OR 2 months after her initial presentation for completion cytoreductive surgery (CRS) and hyperthermic intraperitoneal chemotherapy (HIPEC) with 40 mg of mitomycin-C at a target temperature of 42–43 °C over 90 min, as per our institutional protocol []. Her PCI at the second surgery was calculated to be 13. She had minimal peritoneal adhesions, and her intraabdominal infectious process had completely resolved. The patient underwent an appendectomy, omentectomy, and tumor debulking. The appendicular base was healthy and easily stapled across. The patient was discharged home with lovenox on postoperative day 5, saturating well on room air, tolerating a diet with oral pain medication, and with return of bowel function. Six months after the CRS/HIPEC, the patient had surveillance imaging with no evidence of recurrence (Fig. ). The abdominal wall mass was unchanged from prior imaging and, given the biopsy of leimyoma, presumed to be an implant at the port site from the patient’s prior hysterectomy.\nAfter the discharge from the second surgery, the patient’s postoperative recovery was complicated by new-onset shortness of breath after the completion of her 30 day course of lovenox. She was diagnosed with bilateral pulmonary emboli, with no evidence of deep vein thrombus on lower extremity duplex. Therapeutic lovenox was initiated, and her symptoms gradually improved. An interval angio CT of the chest demonstrated resolution of the clot. Anticoagulation has since been discontinued, and the patient will continue with 6-month interval surveillance imaging for PMP recurrence.\nThe pathology of the left ovary and tube following the first operation resulted as an ovarian cyst containing mucin pools and low-grade intestinal type mucinous glands, consistent with metastasis from appendiceal mucinous neoplasm. The right ovary and tube showed mucin, acute inflammation, and adhesions. Immunostains were positive for CK7, CK20, and CDX2 and negative for PAX8 (Fig. ). Following the completion cytoreduction, the appendiceal pathology demonstrated a low-grade appendiceal mucinous neoplasm (LAMN) with acellular mucin outside the appendix and associated fibroinflammatory response. The proximal margin, the appendicieal base, was negative for tumor. Additional specimens sent during the debulking included the falciform ligament, omentum, pelvic tumor nodules, and tumor deposits overlying the sigmoid, small bowel, left colon, and liver. All additional specimens consisted of acellular mucin. Pre-operative carcinoembryonic antigen (CEA) and CA-125 were elevated at 26.8 and 101 respectively. CA 19-9 was within the normal limits. Using AJCC (8th ed.) staging, the final staging was determined to be T4a NX M1a (stage IVA). Since the patient had a complete cytoreduction and the pathology was determined to be LAMN, no further adjuvant therapy was given. |
The patient was a six-year, 10-month-old male with a past medical history of ADHD diagnosed at five years old who had benefited from extended-release dextroamphetamine prescribed by his pediatrician. Over time, the dose was titrated up to 20 mg daily for the last month prior to evaluation. Side effects had been limited to intermittent abdominal discomfort. He did not take any other medications, nor did he have other significant medical or psychiatric history. There were no complications during pregnancy, delivery, or early childhood. The family composition included his biological parents and two siblings. Family history was significant for anxiety in several family members, a paternal grandfather with schizophrenia, and a younger sister with seizures. No abuse or trauma histories were reported by either the patient or parents. He was seen by his pediatrician within the week prior to presenting to the hospital; collateral information from the pediatrician indicated he was in his normal state of health and his ADHD was well-managed. There was no indication that medications were being mishandled; parents reported that given the age of children in the home, medications were kept well out of reach. Nobody in the home was taking herbal supplements, that if taken by the patient, could have interacted with his dextroamphetamine.\nOn the night of the episode, the patient awoke at three in the morning, then began to describe visual and tactile hallucinations, specifically seeing and feeling spiders in his room, on his body, and in his mouth. He made comments such as “I feel like spiders are crawling on me” and “I feel like I am on fire.” His parents confirmed no spiders were present and attempted to console him, but he became increasingly fearful and agitated. On the way to the Emergency Department, he was screaming without fluent speech, flailing his arms, gesturing to his mouth, and making motions as though to remove bugs from his body. His parents reported he had a preexisting minor insect phobia but was usually easily consoled, and that he had nightmares and night terrors previously but never anything like this episode. His parents felt confident that he was awake and conscious during the episode.\nWhile in the Emergency Department, the patient was persistently agitated, crying inconsolably, and continued to report visual and tactile hallucinations. Though a complete physical and neurological exam was not possible at the time, he was afebrile but tachycardic, tachypneic, hypertensive, and responding to internal stimuli. He received lorazepam 0.5 mg IV several times with little improvement in agitation. Upon transfer to the Pediatric Intensive Care Unit, he was started on dexmedetomidine 0.5 mg/kg/hr IV for sedation that was continued on the first day. At this time, his dextroamphetamine was stopped. For the next two days, he continued to report seeing spiders in his room at times, causing distress. However, these episodes eventually lessened in frequency and severity, and he was once again easily consoled by his parents. After a three-day hospital stay, he was discharged home.\nOn the day after admission, a physical exam revealed a well-appearing, normal stature boy in no acute distress. He had normal vital signs. He had a single café au lait macule over the right nipple. The neurological exam was grossly intact but notable for mild postural tremor of the bilateral upper extremities. Mental status exam revealed a fidgety, anxious, and hypervigilant child worried about his hallucinations returning. He appeared, spoke, and behaved appropriately for his age. As shown in Table , medical workup was largely negative or within normal limits; it consisted of CMP, CBC with differential, ESR, CRP, TSH, T4, ammonia, folate, ceruloplasmin, cortisol, Lyme titers, UA, body fluid culture, COVID-19 PCR, CSF studies, and brain MRI. Urine toxicology was positive for amphetamines, which was expected due to the patient’s prescribed dextroamphetamine. The respiratory pathogen panel was positive for parainfluenza virus type 3. ECG performed on admission showed sinus tachycardia. EEG was not completed in the hospital; the patient would not tolerate it.\nAside from sedation with lorazepam and dexmedetomidine, the patient did not receive medications while hospitalized. His dextroamphetamine was stopped. The patient and his parents were provided with reassurance and were taught coping strategies, deep breathing exercises, and other interventional measures should the hallucinations return. The patient was also scheduled for therapy to be started soon after discharge from the hospital. One month later, the patient restarted his dextroamphetamine as prescribed by his pediatrician. At that time, 24-hour ambulatory EEG did not record any epileptiform activity. Two months after restarting dextroamphetamine, the patient’s hallucinations had not recurred. |
A 35 years old (height, 159 cm; weight, 85 kg; gravida 3, para 1) pregnant woman with SAS presented to our hospital and was scheduled to undergo elective CS under epidural anesthesia at 36 weeks of gestation. An echocardiographic examination at 34 weeks of gestation revealed that the diameter of the beginning portion of the aorta (under the aortic valve) was narrowed by about 17 mm. The diameter of the ascending aorta was normal, the average pressure gradient was 40 mmHg, and the ejection fraction was 67%.\nIn the operation room, her baseline vital signs were as follows: heart rate (HR), 82 bpm with normal sinus rhythm; noninvasive blood pressure (BP), 130/76 mmHg; and oxygen saturation (SpO2), 96%. Supplemental oxygen was administrated by a face mask at the rate of 3 L/min. Traditional epidural puncture was performed at the L1–2 and L3–4 interspaces in the left lateral position, and a catheter was inserted to a depth of 4 cm into the epidural space at the cranial and caudal ends of the surgical field. The patient was then placed supine in a left-tilt position; 3 mL of 2% lidocaine was injected as a test dose, and another 7 mL was injected 5 min later through the cranial catheter. Additionally, 3 mL of 1.5% lidocaine was injected through the caudal catheter. At 20 min after the lidocaine injection, the sensory block had reached T6. The patient had no discomfort, and her hemodynamic parameters were stable. The operating bed was adjusted from the left-tilt to horizontal position, and the operation was allowed. Two minutes later, the patient reported chest distress and difficulty breathing (HR, 110 bpm; BP, 80/69 mmHg; SpO2, 96%), and 3 mg of intravenous ephedrine was promptly administered. The patient immediately lost consciousness with no response (HR and BP were depressed to 40 bpm and 53/15 mmHg, respectively). The lowest BP at the time was not measured because the noninvasive BP measurement interval was set to 1 min. The obstetricians sped up the surgery, and in another 2 min, a 2750-g male newborn was delivered with an Apgar score of 10 at both 1 and 5 min. Spontaneous circulation of the patient was returned immediately after delivery, and stable vital signs returned with no other treatment. The postoperative course was uneventful. The mother and neonate recovered uneventfully and were discharged 4 days later. |
A previously fit and well 18-year-old man presented to the eye casualty with 7 days of increasingly red eyes, no discharge, a feeling of heaviness around the eyes, and oral and penile ulcers. He had been diagnosed with COVID-19 3 weeks earlier and had symptoms of fatigue, anosmia, headache, shortness of breath and sore throat. The eye symptoms and ulcers came on after resolution of his COVID-19 symptoms. On Day 1 of his eye symptoms, he had a remote consultation with a tertiary eye casualty and was given lubricating eye drops for presumed viral conjunctivitis. A day after this, he was prescribed oral penicillin V for a sore throat and developed the penile and mouth ulcers as soon as he took the antibiotics (). His general practitioner (GP) referred him to dermatology and rheumatology for investigation.\nHe was referred for review in eye casualty from the dermatology clinic, and presented 7 days after the red eyes and 6 days after the oral and genital ulceration had begun. On examination visual acuity was 6/6 in the right and 6/9 in the left. There was diffuse injection of the bulbar conjunctiva with bilateral conjunctival pseudomembranes, but no tarsal conjunctival epithelial defect on eversion of the lid (). The corneas were clear with no epithelial defects. He was started on copious lubrication, 2 hourly preservative-free dexamethasone 0.1% drops, and preservative-free chloramphenicol drops 4 times a day as prophylaxis. Two days later, his symptomatology improved, and the bulbar conjunctival injection and pseudomembranes improved significantly. The patient was reviewed again by dermatology and diagnosed with SARS-CoV-2 associated EMM following the results of a 3-mm punch biopsy of the lower lip, which showed ulcerated skin with focal interface mucositis ( and ). He was started on prednisolone 30 mg orally for 1 week. He was advised to continue the chloramphenicol and taper down the dexamethasone drops. Two weeks later he made a full recovery. There were no ophthalmic signs and no remaining ulcers. He was advised to stop the chloramphenicol and taper off the dexamethasone drops gradually. |
A 38-year-old male became unconscious following sudden onset of giddiness. He was admitted to the emergency services of our tertiary care center “in extremis” and died despite resuscitation. He had a history of hypertension, diabetes mellitus, and left hemiparesis although no specific details were available. Apart from a random blood glucose level of 409 mg/dL, routine hematological and biochemical investigations were normal.\nAt autopsy, there was mild cardiomegaly (heart weight 300 gm) with moderate enlargement of the left ventricle. The aorta and pulmonary trunk were normally related although both the trunk and its branches were dilated and thin walled. The ascending aorta was aneurysmally dilated, having a maximum internal diameter of 2.8 cm. The aortic arch was left sided and contained a few calcified atheromatous plaques. The brachiocephalic arteries were similarly mildly atherosclerotic but showed no significant luminal stenosis. Just distal to the origin of the left subclavian artery [LSA, ], however, there was fusiform aneurysmal dilation of the proximal 4 cm of the descending thoracic aorta, which had a diameter of 3.5 cm. Opening the segment revealed occlusive laminated thrombus, with the wall being atherosclerotic and calcified. A distinct constriction was found just beyond the aneurysm, with serial longitudinal sections at this site showing lack of communication with the descending thoracic aorta, a finding confirmed by subsequent microscopy []. The distal segment was supplied by a sprout of four to five thin-walled arteries, one of which was of a large caliber. The origin of these collaterals, however, was unfortunately not noted at autopsy. Our diagnosis was interruption of the aortic arch (IAA) “Type A” interruption.\nFurther examination of the heart revealed bicuspid aortic valve (anterior to posterior type) [], moderate concentric left ventricular hypertrophy, and evidence of spontaneous closure of a preexisting trabecular muscular ventricular septal defect []. The left coronary artery was dominant. All the coronary arteries were diffusely atherosclerotic, with multifocal critical stenosis [Figure and ]. The left anterior descending artery, 3 cm from its origin, showed luminal occlusion for a length of 0.8 cm produced by a fresh organizing thrombus which had developed over an eroded atherosclerotic plaque []. We presume this to be the cause of his sudden cardiovascular collapse. Examination of the brain revealed mild frontal atrophy, atheromatous changes in the vertebrobasilar system and lacunar infarcts in the basal ganglia, thalamus, hippocampus, and pons. There were histological features of hypertensive angiopathy, occasional microaneurysmal formation, and organized thrombus. |
A previously healthy 35-year-old woman was admitted with a 2 weeks history of a crusted ulcer around left eye. The lesion started at outer canthus of the left eye as a painless erythematous pustular lesion with surrounding edema, which later spread in the next 7–8 days to involve upper and lower eyelids along with swelling of the cheek on the left side. There were multiple hemorrhagic bullae that ruptured to form a gangrenous ulcer with an overlying eschar [].\nPatient complained of watering from her left eye along with gradual diminution of vision as a result of orbital swelling obscuring the vision. There was no history of fever before or after the appearance of the lesion.\nThe patient did not give any past history of diabetes mellitus or chronic respiratory illness or any abnormal bleeding. There was no significant drug history, or history of insect bite, or intimate contact with cattle.\nGeneral examination showed no pallor, icterus, cyanosis, clubbing, or pedal edema. The patient was of average build and height with no signs of malnutrition. Pulse, blood pressure, and temperature were within normal limits. No abnormality was detected in the respiratory and cardiovascular systems. Abdomen was soft with normal bowel sounds. The liver, spleen, and lymph nodes were not palpable. Central nervous system examination was within normal limits. The patient was well oriented to time, place, and person.\nLocal examination showed a crusted ulcer situated at the left periorbital region of size 9 cm × 5 cm with irregular shape, ragged margins, raised hyperpigmented border, and floor filled with slough and necrotic tissue. The lesion was nontender and the base was indurated.\nBlood investigations such as complete hemogram, blood sugar, serum urea and creatinine, liver function test, and serum electrolytes were within normal limits. Routine and microscopic examination of urine showed no abnormality. C-reactive protein was within normal range. Serology for human immunodeficiency virus -1 and 2 was nonreactive. Chest radiograph posteroanterior view and ultrasound whole abdomen were normal. Blood culture showed no growth of microorganism. Swab taken from the ulcer showed growth of P. aeruginosa in both blood agar and MacConkey agar media. Punch skin biopsy from the margin of the ulcer stained with hematoxylin and eosin showed necrosis of the epidermis and the upper dermis, covered on the surface by necrotic exudates []. The deeper dermis and subcutaneous fibroadipose tissue also showed necrotic areas surrounded by inflammatory cells with a predominance of polymorphonuclear neutrophils. The epidermis adjacent to the necrotic areas showed hyperkeratosis and acanthosis. No vasculitis was found [].\nSystemic antipseudomonan antibiotic with injection piperacillin and tazobactam 4.5 g thrice daily was started after obtaining the wound swab culture sensitivity report. The lesion completely resolved with subsidence of orbital swelling within 14 days of antibiotic therapy. The patient regained vision in her left eye completely at the end of treatment [].\nConsidering the clinical findings, laboratory investigations and therapeutic response to antipseudomonas antibiotics the case was diagnosed as ecthyma gangrenosum without bacteremia. |
A 46-year-old woman (gravida 2, para 2) was referred to our hospital complaining of a lower abdominal mass and pain. Her medical history was unremarkable. She was initially diagnosed with a uterine leiomyoma by transcervical needle biopsy. CT revealed a large heterogeneous tumor occupying the pelvic cavity and an intravascular tumor within the dilated left internal iliac and ovarian veins (Figures and ). Her preoperative cervical cytology results were negative for intraepithelial lesions and malignancy. The endometrial cytology and needle biopsy results were also negative. Thus, the preoperative diagnosis was IVL, with extension of the tumor into the left internal iliac and ovarian veins.\nIntraoperatively, multiple myomas were found within the uterine corpus and cervix, and the tumor extended to the parametrium and paracolpium. Detachment of the tumor from the left ureter and vaginal wall was very difficult. Intravenous tumors in the left internal iliac and ovarian veins could be palpated. The left internal iliac vein forming the common iliac vein was transected at the bifurcation region. In addition, TAH and bilateral salpingo-oophorectomy (BSO) were performed, resulting in the complete surgical resection of the tumor (operative time, 11 hours; blood loss, 8462 g). The resected uterus and adnexa weighed 897 g (Figures and ). There was no residual tumor detected in the venous resection stump.\nThe nodule resected from the uterus and the internal iliac and ovarian veins consisted of a proliferation of spindle cells. There was no nuclear atypia and the mitotic index was low. In addition, vessel endothelium cells and a vascular smooth muscle layer covered the IVL (Figures and ). The tumor cells stained positive for Alcian blue (pH = 2.5) and the staining disappeared after hyaluronidase digestion. However, compared to that in Case 1, the intensity of the staining was weaker and less diffuse (Figures and ). Similar findings for hyaluronan expression were obtained using the sample retrieved from the preoperative needle biopsy.\nThe histopathological diagnosis of the uterine and intravascular tumors was IVL. There has been no evidence of IVL recurrence, with the most recent follow-up at 38 months postoperatively. |
A one-year-old, previously healthy boy with no history of chronic constipation presented with a five-day history of frequent vomiting, constipation, and abdominal distension. He was 74 cm in height and weighed 9 kg at the time of presentation. Upon examination, he was tachycardic but otherwise relatively stable. His abdomen was considerably distended but nontender and filled with palpable stool, yet a rectal exam revealed no stool. An abdominal X-ray scan demonstrated multifocal air-fluid levels (). Suspecting mechanical obstruction, we performed an abdominal computed tomography scan. It revealed multiple fecalomas in the transverse colon, wall thickening of the descending colon, bowel distension from the jejunum to the transverse colon, moderate ascites, and no evidence of free air (). A gastrografin enema examination revealed expansion failure of the descending colon and the presence of impacted fecalomas in the transverse colon, which could not be resolved by colonic lavage (). His condition was not imminent, and we continued conservative observation. However, the vomiting and inability to pass the stool continued even on the following day, and his abdominal distension had worsened upon nasogastric tube drainage. He was taken to the operating room for an emergency endoscopic fecal disimpaction under general anesthesia.\nA colonoscopy was performed using a scope of the upper gastrointestinal tract (GIF TYPE Q260, 9.2 mm in diameter; Olympus, Tokyo, Japan) with insufflation of carbon dioxide. Edematous mucosa of the descending colon and multiple giant brown fecalomas were observed to be occupying the lumen of the transverse colon (). Although the surface of the fecaloma was hard, large, and slippery, repeated shaving of the fecalomas with biopsy forceps resulted in gradual fragmentation. Finally, after 88 minutes, all fecalomas had been broken into fragments of a size that we thought would be able to pass through the anus. Samples were collected from the colon wall to explore the cause of the situation using biopsy. After endoscopic disimpaction, the patient experienced an intermittent bowel movement immediately, and his abdomen was less distended obviously thereafter. He was able to consume meals after recovery and was successfully dismissed from hospital care 18 days after the endoscopic treatment. Later, the pathology of a biopsy sample taken from the descending colon wall showed 22 eosinophils per high-power field (HPF), while all investigations conducted for intestinal motility disorders such as Hirschsprung's disease were negative (). He was considered to have a food protein-induced enterocolitis syndrome, which is a kind of gastrointestinal food allergy. It was determined that, before the described event, he had eaten barley for the first time in his life. Because an allergy load test is thought to be risky in the context of severe gastrointestinal allergy, we are considering performing the test to confirm his possible barley allergy once he reaches about three years old. |
An 11-year-old female child presented with a swelling in front of neck of 1 year duration; on examination, a solitary nontender globular swelling measuring 5 cm × 4 cm was noted in the submental triangle in front of hyoid bone with well-defined borders [], smooth surface, and nontender, it moved with deglutition and with protrusion of the tongue, thyroid gland was clinically normal, there was no lymphadenopathy, and oral cavity was unremarkable. Provisional diagnosis of the thyroglossal cyst was made. Routine investigations and thyroid function test were within normal limits; ultrasound of the neck showed midline swelling in the infrahyoid region with solid and cystic components with hypoechoic mural nodule and multiple foci of calcifications; thyroid gland was unremarkable. Fine-needle aspiration cytology of the swelling showed features of papillary carcinoma. Contrast-enhanced computed tomography scan revealed a lobulated cystic lesion measuring 55 mm × 44 cm × 35 mm in the anterior neck just below the hyoid bone with solid component showing intense enhancement (170–180 HU) []. With above findings, the patient was planned for Sistrunk procedure. Intraoperatively, there was swelling measuring 5 cm × 4 cm in the submental triangle with a stalk extending to the body of hyoid bone [], and going through it, there was no infiltration of surrounding structures and no obvious lymphadenopathy. Postoperative course was uneventful, drain was removed on the postoperative day (POD) 2, and the patient was discharged on POD3. Histopathological report showed a 5.5 cm × 4 cm × 5 cm swelling with solid and cystic components with multiple small papillary projections; on gross examination, microscopic picture showed papillary carcinoma-classical type [] with lymph vascular invasion, and two lymph nodes were identified of which one showed papillary carcinomatous deposits []. After multidisciplinary discussion, the patient was advised total thyroidectomy and bilateral lymph node dissection, but her parents refused surgery; after 2 years of follow-up, the patient is clinically normal and repeat ultrasound of the neck did not show any abnormality in the thyroid or lymphadenopathy. |
A 32-years-old unmarried woman who had no history of any medical disorder or head trauma and no family history of epilepsy suffering from major depressive disorder had been taking zolpidem for insomnia for more than 1 year. Since one year ago after her brother’s sudden death, she began to take zolpidem alone without mixing other kinds of hypnotics, and 50 mg of zolpidem used to be initially effective in treating her insomnia. In some days, the dose increased up to 100 mg per day About 9 months later, she refers to a psychiatrist because of depression, anhedonia, fatigue, hopelessness and decreased appetite and the psychiatrist prescribed sertraline 50 mg once a day with diagnosis of major depressive disorder, the patient had taken the drugs with no problem for the past 3 months until than she was infected with herpes simplex virus and her doctor prescribed acyclovir 400 mg each 6 hours (1600 mg a day). In the end, she had to discontinue zolpidem abruptly because she could not afford it anymore. After 2 days, she suddenly showed facial spasm, mouth opening, tonic-clonic seizure, and loss of consciousness for about 1-2 minutes. Postictal confusion with clouded consciousness, psycho-motor retardation, persisted in 1 day. So she referred to her psychiatrist and a consultation with a neurologist had been requested by the psychiatrist. The neurological examinations were normal and EEG in wakefulness revealed intermittent, generalized, diffused alpha wave and diffused sharp waves, and suggested seizure waves in patient. As well as we use Naranjo scale for estimating the probability of relationship between seizure and zolpidem withdrawal, the patient score was 10, hence other etiologies or drugs either idiopathic causes were ruled out. After a series of laboratory tests and other examinations, no other etiologies could be identified in MRI. After 4 weeks follow-up, she consumed 5 mg zolpidem at bed time and she had no further seizure attacks and her postictal confusion resolved gradually 1 day after seizure occurred. |
A four-year-old Indian boy presented with a history of swelling and pain over the lumbar spine, low-grade fever, and failure to thrive for 6 months and abdominal distension for 3 months. The abdominal distension had increased since the last 5 days and was associated with a bulge in the left loin. There was a history of an abnormal gait with the child bending forward while walking. There was no weakness in the lower limbs nor were there any bladder symptoms. The mother had received antituberculous therapy for pulmonary tuberculosis 2 years ago. The child had been seen by various private practitioners in his village but was finally brought to Mumbai for investigation.\nPhysical examination revealed an ill-looking pale child. His weight was 11.2 kg and height 89 cm. both below the 5th percentile for age. There were 3 left inguinal lymph nodes, matted and nontender, the largest of them being 2 cm by 3 cm in size. There was a gibbus at the level of L4-L5 vertebrae. The abdomen was distended with palpable bilateral large cystic masses. The left-sided mass was 20 cm by 10 cm, whereas the one on the right side was 15 cm by 10 cm in size. There was also a swelling in the left loin which was 6 cm by 8 cm in size ().\nCentral nervous system examination revealed a conscious child with normal higher functions and cranial nerves. Examination of the motor system revealed normal tone and power in both upper and lower limbs. The deep tendon reflexes were brisk in both lower limbs. The plantar reflexes were flexor bilaterally. Abdominal and cremasteric reflexes could be elicited normally. The sensory system was normal.\nInvestigations showed hemoglobin of 7.1G/dL, a total WBC count of 15,000/cu·mm with a differential count of 25% lymphocytes and 75% polymorphs, and a platelet count of 6.4 × 105/cu·mm. The erythrocyte sedimentation rate (ESR) was 80 mm at the end of 1 hour. The ELISA for HIV was negative. Lateral X-ray of the lumbosacral spine showed a wedge-shaped collapse of L4 vertebral body with bony fragments in the prevertebral space (). Ultrasonography (USG) of the abdomen revealed bilateral large psoas abscesses with extension of the left psoas abscess through the left paraspinal muscle into the subcutaneous plane posteriorly. The computerized tomography (CT) scan of the abdomen () showed lesions of cystic density in both psoas muscles with peripheral enhancing walls and calcific areas within. There was destructive collapse of L4 vertebra with peri- and paravertebral abscesses communicating with the psoas abscess bilaterally. There was also an epidural abscess at L4-L5 level compressing the spinal cord. There was partial rotation of the right kidney with the pelvicalyceal system facing anteriorly.\nUltrasonography-guided aspiration was undertaken on both sides using pigtail catheters and the drains were kept in situ. Thick white pus was drained (150 mL on the left side and 50 mL from the right side). Staining of the pus and culture for acid-fast bacilli was positive. Routine bacterial culture was sterile.\nThe patient was immobilized and started on antituberculous therapy with isoniazid (5 mg/kg/day), rifampicin (10 mg/kg/day), pyrazinamide (25 mg/kg/day), and ethambutol (20 mg/kg/day). Intravenous antibiotics ceftriaxone and amikacin were also started pending the report of bacterial culture. Three weeks later the USG showed almost complete resolution of the abscesses and the drains were removed. An external lumbar brace was provided to facilitate ambulation. With the above treatment, the patient showed a steady weight gain with decrease in the abdominal distention. At discharge on day 30, the child was afebrile, weighed 14 kg, and had no neurological deficit. At followup a month later, the child was gaining weight and had a normal gait. |
A 36-year-old man who denied previous systemic disease had a history of drug abuse with ketamine for 6 to 7 years (at a frequency of 2–3 times per week, by nasal inhalation, and hence the dosage could not be measured), and had then ceased use for approximately 4 years.\nHe had suffered from dysuria, bladder pain, and a mild burning sensation during urination, especially over the urethral meatus and the perineal region, for approximately 1 month prior to admission. He ignored these symptoms initially, but the burning pain worsened, with concomitant urinary frequency and urgency. He visited another hospital for help, at which routine urine analysis revealed pyuria. Under the impression of acute prostatitis, oral antibiotic treatment with ciprofloxacin was initiated during an outpatient visit; however, his symptoms remained, with no improvement. Two days before admission to our hospital, the symptoms worsened, with a newly-developed decreased voiding amount (approximately 50 mL per void) and urgency with urge incontinence, accompanied by painful hematuria and blood clot formation, especially at the first urine void of the morning. The patient then presented to our Emergency Department. Urine analysis showed pyuria, over 100 white blood cells (WBCs)/high-power field (HPF), and significant tenderness and swelling of the prostate was noted upon digital rectal examination, but no pus-like urethral discharge was seen. Under the impression of acute prostatitis, for which oral antibiotic treatment had failed, the patient was then advised to undergo hospital admission for advanced antibiotic treatment.\nAfter admission, we consulted an infectious disease specialist for evaluation, and antibiotic treatment with ceftriaxone was started immediately. A blood test revealed WBC 4870/μL without predominance of neutrophils or eosinophils. No marked elevation of serum C-reactive protein (CRP) (0.25 mg/dL) was noted. The patient's temperature after admission had remained within the normal range, and there were no accompanying signs or symptoms of toxicity. Several blood and urine cultures were performed, including tuberculosis, but all results were negative. The symptoms of pyuria (which remained over 100 WBCs/HPF), urgency, and painful hematuria persisted with no improvement after one week of intravenous antibiotic treatment. However, a sonogram performed upon admission revealed suspected bladder wall thickening. Due to the persistent symptoms that failed to respond to advanced intravenous antibiotic treatment for 1 week, abdominal computed tomography (CT) with contrast was then arranged. The CT scan showed asymmetrical wall thickening (thickness of up to 1.2 cm) of the anterior aspect of the urinary bladder with a mural nodule, and mucosal enhancement with perivesical fatty stranding (Fig. ). According to the above findings and the clinical symptoms, bladder cancer was highly suspected, and we discussed cystoscopy with bladder biopsy with the patient and his wife, which was then performed the next day. Prior to hydrodistension, the bladder mucosa presented with hypervascularity, but there was no tumor over the anterior wall of the bladder as seen on the CT scan. The bladder mucosa of the anterior wall was erythematous, with multiple hump-like changes, and several biopsies were performed. After hydrodistension at a pressure of 90 cm H2O for 8 minutes, the bladder capacity was approximately 150 mL, and bleeding over multiple aspects of the bladder was seen, with glomerulation and ulcerative changes (Fig. A and B). Pathologic analysis of the bladder biopsies showed erosive cystitis, characterized by denuded urothelial cells, with prominent infiltration by eosinophils, lymphocytes, neutrophils, and plasma cells over the mucosa and submucosal layer. In addition, hypervascularity and submucosal granulation formation with fibrosis were observed (Fig. A and B).\nAfter the operation, the bladder capacity increased a little, to approximately 70 to 80 mL per urination, but the urgency, frequency, nocturia, and hematuria still persisted. We also prescribed an anticholinergic agent, a beta-3 agonist and a nonsteroidal anti-inflammatory drug (NSAID), and the patient was then discharged. During 2 weeks of outpatient treatment, his symptoms did not improve with medication. Thus, we discontinued medical treatment and performed hyaluronic acid (HA) instillation, once a week for a total of 10 times. After the treatment, the symptoms of urgency, frequency and nocturia improved, and the bladder capacity increased to 350 mL per urination according to the patient's own voiding diary; in addition, no morning hematuria or hematuria after holding back urine occurred. After the patient's symptoms had improved, we arranged follow-up MRI of the bladder and cystoscopy; on the images, no thickening of the bladder wall nor nodules were observed (Fig. ). Cystoscopy showed marked improvement of the previously-noted erythematous bladder mucosa, and there was neither active bleeding nor glomerulation seen during the whole procedure. Bladder biopsy near the previous biopsy site was performed, and the final pathologic analysis showed decreased inflammatory cell infiltration, regeneration of the urothelium, and less vascularity (Fig. A and B).\nWritten informed consent to publish this case report was provided by the patient, and the consent procedure was approved by the Ethics Committee of Tri-Service General Hospital. |
An 11-year-old boy was injured after falling to the floor and landing on his right hand. He complained of severe pain in his dominant right elbow. He came to our hospital on the same day. The initial examination showed severe swelling on the medial side of his right elbow and pain. The initial X-ray studies of the elbow showed a fracture of the medial condyle of humerus. The bone fragment was displaced laterally by the traction of the flexor muscles; however, dislocation of the humeroulnar joint was not observed (). We diagnosed the patient with a medial condyle fracture of the humerus (Salter–Harris [] type IV, Milch type [] I, and Kilfoyle type [] III). On the next day, open reduction and internal fixation of the fracture were performed under general anesthesia. An incision was made over the fragment. First, the ulnar nerve was identified (). The reduced bone fragment was fixed with three Kirschner wires in the accurate position. Osteosynthesis with a compression screw would have been the optimal treatment. Since multiple temporary fixations with wires were needed to fix the unstable bone fragment, we were afraid of bursting the fragment with a compression screw ().\nA long arm splint was applied from the upper arm to the metacarpophalangeal joints, with the forearm kept in a neutral position for four weeks.\nThe three Kirschner wires were removed at 8 weeks after surgery under general anesthesia (). Ulnar nerve palsy was not noted during the follow-up period. At 1 year after surgery, the absorption of the trochlear groove was observed on an anteroposterior view radiograph of the elbow and thus demonstrated a so-called “fishtail deformity.” However, the patient did not complain of elbow pain, and the range of motion was not limited.\nThe absorption of the trochlear groove was gradually remodeled before the most recent follow-up examination (at four years after surgery). At the latest follow-up examination, a good range of motion was observed, with 0 degrees of extension and 140 degrees of flexion.\nThe patient and the patient's parents were informed that this case study would be submitted for publication and provided their informed consent. |
A 24-year-old female with a history of uncomplicated Crohn's ileitis since the age of 18 presented with right flank pain of 3 weeks' duration. She was 31 weeks pregnant. She had been in clinical remission on azathioprine maintenance therapy for her Crohn's ileitis. However, she elected to discontinue her azathioprine during her pregnancy. Physical examination was significant for tenderness in her right flank and limitation of range of motion due to pain in her right lower extremity. Magnetic resonance imaging (MRI) showed a right iliacus muscle abscess 7 × 5 cm (Figures and ). Ultrasound-guided aspiration of the iliacus muscle abscess yielded 90 mL of purulent fluid. Cultures grew multiple enteric organisms. She responded to 4 weeks of intravenous antibiotics and percutaneous drainage, with resolution of the abscess by MRI (). She delivered a healthy baby at 37 weeks of gestation by vaginal delivery after induction of labor. Two months postpartum, she complained of recurrence of her right flank discomfort. She had no fever or chills. Laboratory examination was normal, without leukocytosis. Computerized tomography revealed that the iliacus muscle fluid collection had recurred. There was no visible fistulous communication from the thickened ileum to the right iliacus muscle. Percutaneous aspiration revealed scattered white blood cells, but no organisms on gram stain, fungal stain, or culture. The sterile fluid collection was treated by percutaneous drainage until resolution, and then the drain was removed. She underwent ileal resection. No residual abscess or fistula was identified at surgery. One month postoperatively she again complained of right flank pain. She had no fever or leukocytosis. Imaging studies () revealed that the iliacus muscle fluid collection had again recurred. Percutaneous aspiration again revealed a sterile fluid collection. An abscessogram () outlined the seroma cavity and excluded an ongoing fistula. She responded to a prolonged course of percutaneous drainage and eventual sclerotherapy of the residual seroma cavity. |
A 50 year old female was referred to our outpatient clinic for evaluation of an allergic reaction to metronidazole. Within 20 min of receiving a therapeutic dose of metronidazole 500 mg to treat a Trichomonas vaginalis infection, she had experienced hives and throat swelling, which resolved with diphenhydramine. She had similarly reacted years prior, when she was prescribed metronidazole for an episode of diverticulitis. Past medical history was relatively unremarkable, though she did note diverticulitis, hypertension, fibromyalgia, chronic fatigue, and a previous hysterectomy. Concomitant chronic medications included amlodipine, perindopril, and boric acid. She had a history of allergic reaction to ranitidine, with documented reaction of throat tightness and hives. The patient was experiencing significant discomfort from her infection, and after discussion of the risks and benefits in our clinic, elected to undergo desensitization to metronidazole accordingly.\nThe modified oral desensitization protocol adapted by Grendelman et al. in 2014 was undertaken. We collaborated with a local community compounding pharmacy, and asked for their help in providing compounded suspensions of oral metronidazole to undertake the desensitization protocol. The compounding pharmacy provided a suspension of oral metronidazole 50 mg/mL, which was further diluted down prior to administration. To obtain the smallest doses early on in the protocol (Table ), small volumes were diluted serially. Doses were administered in 15 min increments. After the first dose of 0.0025 mg, our patient reacted. Accordingly, Grendelman et al.’s protocol was further adapted for slower dose escalation (Table ). Dilutions were calculated in writing prior to administration of each dose, to ensure mathematical accuracy. Our patient tolerated the escalating doses on day 1 until she reached a dose of 250 mg, at which point she developed objective hives without other systemic symptoms of anaphylaxis. She was given bilastine 20 mg and monitored until her hives resolved, and was provided a prescription for an adrenaline auto-injector to carry overnight.\nShe returned the following day for further desensitization. Her hives went away the preceding evening. She reported some non-specific back pain. She had been provided with a tablet of bilastine 20 mg to take prior to returning on day 2. On day 2, she was given 250 mg and then 500 mg of metronidazole, from the original 50 mg/mL suspension obtained from the pharmacy, and monitored. She did not experience any reaction or discomfort. She was instructed to carry the adrenaline auto-injector with her for the week, and to complete a 7 day course of metronidazole 500 mg twice daily without interruption. She did this successfully, and her infection was eradicated. |
A 38-year-old male presented to the hospital with a chief complaint of abdominal pain and hematuria 1 day after eating a barbecue pulled pork sandwich. He described the pain as ‘deep’ in his abdomen, sharp and non-radiating with no episodes of nausea, emesis or diarrhea. He denied any dysuria or frequency. The patient's past medical and surgical history was unremarkable. He had no abnormalities on his laboratory values besides a urinalysis demonstrating 2+ blood and 11–24 RBCs/hpf. Physical examination revealed point tenderness to deep palpation to the right of his umbilicus, but his abdomen was soft with no signs of peritonitis. An upright abdominal X-ray was unremarkable; however, a CT of his abdomen demonstrated a 2-cm linear density representing the FB in the posterior third portion of the duodenum perforating into the retroperitoneum abutting the right ureter causing hydronephrosis (Figs and , arrow). Two separate attempts were made at endoscopic retrieval in the first 24 h without successful visualization or removal of the FB. Due to the ureteral involvement and hydronephrosis, an operative intervention was recommended.\nUrology performed a right retrograde pyelogram demonstrating a point of obstruction in the proximal ureter with no contrast extravasation. A guide wire and stent were passed through the obstruction into the hydronephrotic right kidney without difficulty. Following successful stent insertion, fluoroscopy was used to confirm the location of the FB at the junction of the second and third portion of the duodenum. A full Kocher maneuver was performed and the duodenum and ureter were separated. A small, linear, FB was identified projecting from the posterior third portion of the duodenum into the ureter (Fig. ) and extracted without injury (Fig. ). The patient's original abdominal pain resolved after the surgery. He was able to tolerate a diet and be discharged on postoperative day 4.\nPathology confirmed a wire metal bristle, likely from a grill brush. |
This case study describes a 5-year-old boy diagnosed with severe and persistent ITW. No risk factors were noted in his perinatal history, and there was no family history of ITW. His psychomotor development proceeded appropriately until the age of 2 years when he started to walk with a toe-walking gait pattern. ITW persisted until he was 4 years of age. Specifically, there was no evidence of increased muscle tone or altered reflexes, and there appeared to be no neuromuscular explanation for the toe walking. The patient exhibited no evidence of foot abnormalities, and the absence of neuromuscular disease, confirmed by magnetic resonance imaging, revealed the clinical diagnosis of ITW. Importantly, although this patient underwent 1 year of physical therapy (PT), including home exercises and nighttime bracing with plastic orthotics to maintain the stretch/flexibility of the Achilles’ tendons, his toe-walking condition worsened over time. In the opinion of an orthopedist, these developments were sufficiently severe to warrant corrective osteotomy, but the patient’s parents refused to provide consent. In this case, a 1-year, nonsurgical treatment program, including TICs combined with a PT program based on NDT principles, was offered and accepted by the parents of the patient.\nWith both knees in extension, he did not exhibit a passive aDF to neutral plantigrade. Upon clinical evaluation, hyperactivity of the toe-grasping reflex could be observed, which intensified with the placement of the subject in the upright position. The parents estimated that the patient spent 100% of his time on his toes. In addition, bilateral Achilles’ tendon contracture developed, both forefeet were splayed, and a disproportionately wide forefoot was observed compared with the heel and external tibial torsion, which frequently develops to compensate for the lack of a flat foot contact ().\nWritten informed consent was obtained from the parents of the patient for the publication of this case report and any accompanying images. A copy of the written consent will be made available for review by the Editor-in-Chief of this journal. The Bioethical Committee of the Medical University of Silesia in Katowice, Poland approved the current report. |
The patient is a 71-year-old white male who was found to have a 3.5 cm right kidney mass and had been followed by the urology team closely at VA Pittsburgh Healthcare System. Urine cytology was suspicious for malignant cells. He underwent a radical right nephrectomy on February 3, 2014. Pathology showed clear cell RCC. The tumor was located at the lower pole with a size of 4.5 cm (pT1b) and Fuhrman nuclear grade 2. All margins were not involved by carcinoma, and there was no vascular invasion. He had been followed with a regular CT scan every year. He was found to have small bilateral lung metastasis and lymphadenopathy in 2016. The PET scan on April 26, 2016, revealed FDG activity in the lung and hilar and mediastinal lymph nodes. He underwent endobronchial ultrasound biopsy of the mediastinal lymph node which confirmed to be metastatic from clear cell RCC. Due to his comorbidities and mild thrombocytopenia, we started him on lower dose sunitinib at 37.5 mg per oral daily ×4 weeks every 6 weeks in May 2016. In total, he received 7 cycles of sunitinib. He had been followed every 6 weeks in the clinic. He only developed fatigue due to mild hypothyroidism for which he received levothyroxine. During the follow-up, he was found to have worsening thrombocytopenia with platelet counts in the range of 60,000 to 90,000. A follow-up CT scan and PET scan in October 2016 showed improvement of the lung metastasis and lymphadenopathy. He was last seen in the clinic on March 13, 2017.\nHe was admitted on March 29, 2017, due to muscle weakness, fatigue, poor oral intake, and difficulty swallowing for 2 weeks. During admission, his platelet count was found to be 13,000, serum creatinine 2.3, total bilirubin 4, AST/ALT > 2000, INR 2.9, calcium 7.5, creatine phosphokinase (CPK) > 5000, and uric acid 12 (see ). Sunitinib was discontinued on the first day of admission. CT head revealed no evidence of metastatic disease. Chest X-ray did not show evidence of infiltration or effusion. Echocardiogram showed severe global hypokinesia with LVEF of 30–35%. His LVEF was 55% prior to starting on sunitinib. He quickly developed lactic acidosis and acute respiratory failure. In the intensive care unit, he received bicarbonate, high-dose oxygen, furosemide, and treatment for hyperkalemia. Despite all treatment support, he continued to decline. His family chose to deescalate care, and he died on April 1, 2017. |
A 45 year-old woman of Caucasian origin presented to the surgeon with a 6-month history of light pain in the upper third of the abdominal wall. In her medical history, the patient had undergone surgery for tonsillectomy, appendectomy and carpal tunnel syndrome and was suffering from arterial hypertension and thyroid disease, treated with ACE inhibitor and levothyroxine sodium.\nUltrasonography (10 MHz probe), performed in May 2013, showed a 20 mm oval formation localized in the right rectus abdominis muscle with inhomogeneous echogenicity and internal vascular signals.\nComputed Tomography (CT) scan with contrast, performed in August 2013, showed inhomogeneous solid nodule with maximum transverse diameter of about 16 mm and a longitudinal extension of 30 mm localized in the right rectus abdominis muscle and characterized by moderate contrast enhancement, without clear demarcation on the profile and light anterior extension in the intra-abdominal adipose tissue.\nContrast magnetic resonance imaging, performed in October 2013, displayed a 20 mm oval formation, localized in the right rectus abdominis muscle, hypointense in sequences T1-T2-weighted, homogeneously hyperdense in the late phase with subcutaneous outer profile and intra-abdominal inner profile, without signs of infiltration of the underlying peritoneum (Fig. ).\nPhysical examination revealed an oval shaped swelling, motionless on the surface and deep levels, with hard consistency and slightly irregular margins.\nAt superficial palpation there were no signs of pain; deep palpation evoked moderate soreness at the epigastrium.\nIn November 2013, the patient underwent to surgical removal of the lesion: surgical access through midline incision of epigastric region showed a not perfectly round hard mass, with adherence zone to the fibers of the right rectus muscle; en block excision was performed with the simultaneous removal of subcutaneous tissue, muscle fascia and parietal peritoneum. No early complication occurred.\nMacroscopically the surgical specimen was composed of yellow-brown tissue. The cut surface revealed a nodular yellow-grey lesion, with irregular borders and 3 cm of diameter. Microscopically, the histological examination of entire surgical specimen showed a proliferation of cellular elements arranged in chains and in nodular aggregates, characterized by large granular, slightly eosinophilic cytoplasm and small eccentric nuclei. These cellular elements appeared interspersed in dense fibrous stroma and accompanied by multiple nodular foci of lymphoid infiltrate. Marginally, the lesion was in continuity with striated muscle and adipose tissue, so it resulted completely excised (Fig. ).\nImmunohystochemical analysis of the above mentioned cellular elements showed positivity for Vimentin (clone V9 Novocastra Leica Biosystems), S-100 protein (clone S1/61/69 Novocastra Leica Biosystems) and CD68 (clone 514H12 Novocastra Leica Biosystems), negativity for alpha-Smooth Muscle Actin (clone asm-1 Novocastra Leica Biosystems), Muscle Specific Actin (clone HHF35 Novocastra Leica Biosystems), Desmin (clone DE-R-11 Novocastra Leica Biosystems) and CD34 (clone QBEND/10 Novocastra Leica Biosystems); the proliferative index (Ki-67, clone MM1 Novocastra Leica Biosystems) was less than 1 % (Fig. ).\nElectron microscopy showed skeletal muscle tissue surrounded by neoplastic cells that show a high number of intracytoplasmic granules of various sizes containing glycogen (Fig. ).\nAccording to these morphological and immunophenotypic characteristics, we made the diagnosis of intramuscular granular cell tumor. |
A 46-year-old female patient was brought to our Accident and Emergency Department with a 5-day history of a painful perianal and perineal swelling, which was increasing in size each day and discharging pus. This was preceded by perineal scratching for itchiness. A day prior to presentation, the pain had increased in intensity and the patient had difficulty in passing stool.\nShe had asthma and hypertension, which were well controlled. She was on salbutamol 4 mg three times a day and used becotide inhaler for her asthma and nifedipine 20 mg twice a day for her blood pressure. Her recent human immunodeficiency virus (HIV) test was negative. There was no family history of diabetes or malignancies. She was married with one child, and she neither drank alcohol nor smoked cigarettes.\nOn examination, she was ill-looking and in pain. She was hypotensive with a blood pressure of 85/35 mmHg, a pulse rate of 110 beats per minute, and respiratory rate of 30 breaths per minute. She had an elevated temperature of 38 degrees Celsius and a low oxygen saturation of 88%. Examination of her perineum and perianal area showed a huge mass with extensive necrosis and oozing pus from its surface. This is shown in her preoperative image below (Figure ).\nCrepitus was elicited in the tissues adjacent to the ulcer. There was exquisite tenderness on digital rectal examination. Examination of other systems was normal. A random blood sugar test done showed a high blood sugar of 18.05 mmol/L. A diagnosis of necrotizing fasciitis was made in a patient with newly diagnosed diabetes mellitus.\nThe patient was very sick and in septic shock from fulminant sepsis. She needed high dependency unit (HDU) admission for septic shock treatment before surgery. Aggressive fluid resuscitation, antibiotic treatment, and insulin per sliding scale for glycemic control were commenced. Empirical antibiotics used were ceftriaxone 1g daily and metronidazole 500 mg intravenous 8 hourly. Her preoperative investigations are shown in Table below. She had a markedly elevated white cell count. After 48 hours of resuscitation and monitoring, the patient's blood pressure improved to an average of 120/80 mmHg. Pulse and temperature came down to 100 beats per minute and 37.6 degrees Celsius, respectively. The patient was taken to theater for debridement and diverting loop colostomy as shown in Figure and B, respectively. Specimens were taken for microscopy, culture, and sensitivity at the time of surgical debridement. Her preoperative investigations are shown in table below (Table ).\nPostsurgery, the patient was admitted in HDU for continued fluid resuscitation, antibiotic, analgesic, and oxygen therapy. Due to good progress, she was discharged to the surgical ward on day 3. The microbiology results grew coagulase-negative Staphylococcus aureus sensitive to vancomycin and Klebsiella pneumoniae sensitive to amikacin. She was commenced on vancomycin 500 mg and amikacin 300 mg intravenous 8 hourly. Treatment with negative pressure wound therapy (NPWT) or vacuum-assisted cure (VAC) was too expensive for the patient. As an alternate to wound care, she had salt sitz baths twice a day followed by dressings with silver sulfadiazine. We noted a positive correlation between rising hemoglobin (Hb) levels, decreasing white cell count (Wcc), and glycemic control and wound size as shown in Figure . By week 4, the wound had spontaneously closed and the patient did not need operative wound closure.\nAt 8 weeks, she had an uneventful reversal of colostomy. She takes glibenclamide 5 mg twice a day for her diabetes in addition to diabetic diet. She has since been seen twice in the surgical outpatient department and did not have any complaints. She has been put under the care of physicians for her diabetes, asthma, and hypertension. The figure below (Figure ) shows reduction of wound size and improvement of Wcc, Hb, and blood sugar levels. |
A 33-year-old woman visited the Department of Advanced General Dentistry at the Dankook University College of Dentistry for third molar removal in 2020. She had no medical history, and was not on any medication. Radiographic evaluation showed that the left lower third molar was impacted and very close to the inferior alveolar canal (). After informed consent, surgical extraction of lower third molar was performed under IAN block anesthesia using 2% lidocaine with 1:100000 epinephrine (Huons, Sungnamsi, Korea). No intraoperative complications occurred, and the IAN was not visualized during the surgery.\nThe next day, the patient revisited, complaining of an altered sensation on the left side. The extraction site was not painful, but she felt dullness and swelling in the left half of the lower lip and chin area. She also complained of increased sensitivity of the left lower central and lateral incisors to cold stimuli. On clinical examination, paresthesia was observed from the vermilion border of the lip down to the chin, but no edema or redness was observed in the affected area (). Intraoral examination revealed slight swelling and redness of the extraction site, and tenderness in the submandibular area. The lower anterior teeth had slight crowding, but there was no gingival edema or redness around the teeth (). On radiographic examination, no periapical lesion or alveolar bone loss was observed in the lower anterior teeth (). The postoperative cone-beam computed tomography revealed a slight discontinuity of upper cortical layer of the inferior alveolar canal ().\nA diagnosis of neurosensory problems of the IAN after third molar removal was made, and neurophysiologic quantitative sensory testing [] was performed. To evaluate sensory function, objective symptom tests (brush stroke, pinprick, and two-point discrimination) were performed, and the patient was asked to compare the level of sensory perception on the affected side with that on the unaffected side using a 10-point numeric rating scale (relative affected side score when unaffected side sensation is 10). A cotton swab was gently brushed over the chin area for the assessment of light touch (brush test). A dental explorer was gently pressed on the chin to indent the skin for sharp pain or a pricking sensation (pinprick test). A compass with blunt tips was used to measure the minimum distance between two points that the patient could distinguish on the affected and unaffected sides (two-point discrimination). There was no difference between the left and right sides in the pinprick and two-point discrimination tests, but the left side showed a lower score than the right side in the brush test (). I performed tooth sensitivity test on cervical area of the lower anterior teeth using an ice stick. The left lower central and lateral incisors had a higher aching response (hypersensitivity) than the adjacent teeth. The response to the percussion test was normal, indicating no inflammation of the periodontal ligament (). I diagnosed hypoesthesia (paresthesia) of the chin and hyperesthesia (hypersensitivity) of the lower anterior teeth after IAN injury.\nTo control inflammatory reactions in the injured nerve and improve nerve regeneration, I prescribed steroids, anti-inflammatory drugs, and Vitamin B12. A 30 mg dose (6 tablets of 5 mg) of steroids (Solondo®, Yuhan, Korea) was prescribed, and the patient was instructed to decrease the dose by 5 mg daily. Ibuprofen 400 mg and Vitamin 0.5 mg (M-Cobal®, Donghwa, Korea) were prescribed three times daily for 2 week. The patient returned to the clinic two weeks later. The tooth hypersensitivity had disappeared, and the sensory dullness of the lips and chin area decreased. At the follow-up one month later (6 weeks after extraction), the paresthesia of the chin had completely resolved. |
A 24 year old man presented with stupor, mutism and food refusal. He had a negative personal and family history of mental illness until four years prior to admission. At that time, at the age of 20 years old, he was first hospitalized after being found wandering dazed in the streets. Diagnosed with schizophrenia, catatonic type, he received haloperidol initially but was switched to olanzapine and lorazepam. He recovered and returned home, until about twelve months later when he was again admitted to a local hospital for two months with profound stupor, immobility and inanition. He recovered on a combination of haloperidol and lorazepam.\nAs an outpatient, haloperidol was switched to olanzapine, but both olanzapine and lorazepam were subsequently discontinued due to side effects of weight gain and sedation, resulting in a recurrence of catatonia and re-hospitalization about ten months after his last discharge. During a complicated admission lasting four months, he underwent sequential trials of olanzapine, risperidone, and lorazepam without any improvement in catatonic symptoms. A trial of electroconvulsive therapy (ECT) was initiated, but despite receiving ten bilateral brief-pulse treatments over one month with an average stimulus energy dosage of 386.2 millicoulombs resulting in ten seizures lasting an average of 37.9 seconds, he showed no change or improvement in his catatonic symptoms and remained stuporous. He finally recovered on a combination of parenteral haloperidol and lorazepam, and was discharged on oral lorazepam and risperidone with monthly paliperidone injections due to suspected medication non-adherence.\nAfter discharge, he showed no signs of residual psychosis or mood disorder for over eight months and was able to return to employment. During remission, his speech was spontaneous, he showed greater emotional expressiveness and he had no psychomotor retardation. However, his affect was mildly blunted and he had limited insight into his illness. Due to his complaint of lethargy and the possibility that some of his residual symptoms represented drug-related extrapyramidal side effects, oral risperidone and lorazepm were discontinued over the next six months, at which time an acute catatonic episode recurred and he was admitted to our hospital.\nOn this index admission, he was mute and supine with few spontaneous movements. He had mild cog-wheel rigidity without waxy flexibility or catalepsy. His affect was flat and expressionless. Automatic obedience could be elicited by passively moving his limbs into new positions despite verbal instructions to resist. Stereotypies, mannerisms, grimacing, ambitendency, verbigeration, and echo phenomena were absent. He showed negativistic behavior and was rated 21 on the Bush-Francis catatonia rating scale (BFCRS).\nExtensive laboratory investigations, including serum chemistries, hematology, serology, toxicology, cerebrospinal fluid examination, neuroimaging and electroencephalography, revealed no significant abnormality.\nParenteral lorazepam was titrated up to 10 mg daily, but with no response after two weeks, haloperidol 7.5 mg was injected twice daily with diphenhydramine 50 mg. Within one week, full remission was achieved with a BFCRS score of zero. After recovery, he was able to speak spontaneously and he again denied any delusions, hallucinations, or changes in mood during or between catatonic episodes. After discharge, he showed mild psychomotor slowing, affective flattening, and limited insight into his illness, similar to previous remissions, but was able to resume employment. |
A 33-year-old married woman, gravida 4 para 3 (three previous caesarean sections), with no relevant medical or obstetrical history, presented to the emergency room at 36 weeks of pregnancy with severe abdominal pain. Until that point, the course of the pregnancy was uneventful without any history of abnormal placentation or hypertensive disorder.\nThe patient complained about acute abdominal pain that started 20 min just after vaginal intercourse. Physical examination revealed marked pallor, abdominal tenderness without uterine rigidity and vaginal bleeding. The cervix was long and posterior. The patient denies any history of direct trauma or domestic violence and there was not any sign of trauma on the clinical exam. Her blood pressure was 90/50 mmHg, the pulse rate 68 beats per minute (bpm). The nonstress test (NST) revealed a normal fetal heart rate (145/min) with moderate variability and no uterine contractions were detected. The patient responded briefly to the rapid infusion of 1 l of normal saline before she collapsed and re-experienced hypotension (80/50). A bedside ultrasound, that was performed in the labor room, revealed a hemoperitoneum and fluid in the Morison pouch. The NST showed a prolonged deceleration for 5 min. An emergent caesarean delivery was decided.\nLaboratory results showed hemoglobin of 8.4 g/l; platelets, coagulation panel, fibrinogenemia, liver function test, and lipase, were all normal.\nPfannenstiel incision was done under spinal anesthesia. Intra-abdominal adhesions caused by previous cesarean sections were noted. A large hemoperitoneum containing more than 2 l of fresh blood and clots was noted. A transverse incision on the lower uterine segment was done and the baby was extracted. We did not identify any abnormality of the uterus or the placenta neither a retroplacental hematoma. The uterine veins and adnexa were normal. However, the bleeding continued and we remark that it was coming from the upper abdomen.\nThe abdominal incision was extended vertically along with general anesthesia. Exploration showed intra-abdominal adhesions and a double tear in the spleen was seen on the splenocolic and gastrosplenic ligament insertions showing active bleeding (). Conservative treatment of the spleen was performed by a general surgeon using hemostatic sutures and compression. During the surgery, the patient received a total of seven units (three packed red blood cells, four fresh frozen plasma). Her post operative hemoglobin was 7 g/l and she received two additional packed red blood cells. The patient's postoperative course was uneventful, and she was discharged home on day 5.\nOf note, the decision-to-delivery interval was 20 min during which the fetal heart rate was normal (category I) except for the last 5 min of deceleration, just before the transfer to the operating room.\nWhen delivered, the newborn presented an Apgar score = 0. The infant was resuscitated at birth and then intubated. Apgar scores were 0/3/4 at 0/5/10 min, respectively. He was admitted to the neonatal intensive care unit (NICU) where umbilical cord blood gas showed severe acidemia (pH: 6.68). His hospitalization was complicated by a resolutive episode of acute renal failure. Cultures were negative. He was extubated on day 4 and feeding started progressively with good swallowing reflex. After six months, the baby presented a normal neurological development in his follow-up visit.\nAn electroencephalogram was done and was not conclusive. He was discharged on day 18. |
A 62-year-old female patient was referred by her oncologist for a routine dental evaluation. The patient’s medical history revealed a cutaneous melanoma of the left dorsum diagnosed six years before she was referred to our dental service. The tumor was primarily treated by surgery and developed two local recurrences (one and three years following surgery, respectively), which were also managed by surgical resection. Disease progression was identified five years after the first treatment and confirmed by a computed tomography (CT), which revealed multiple organ involvement including the lungs, skin (subcutaneous nodules on the dorsum) and bone (osteolytic lesions in the iliac and femur bones, ribs, vertebrae, sternum and scapula).\nThe patient was undergoing a palliative treatment protocol based on dacarbazine, zoledronic acid and radiotherapy in the lumbar region (total dose of 20 Gy) and in the left supraclavicular fossa (total dose of 36 Gy) when she was referred to our dental facility. An extraoral clinical examination identified a pigmented subcutaneous nodule on the patient’s dorsum, measuring 5 cm in diameter, and scarring from the previous surgical resections.\nThe patient’s chief complaint was the loss of the dental crown of her upper left incisor. She denied oral pain or the existence of any relevant oral soft tissue lesion. Intraoral soft tissue examination revealed a nodule with an ulcerated surface and areas of telangiectasia, on the posterior left lateral border of the tongue, measuring approximately 1 cm in diameter, adjacent to a partial edentulous mandibular area (Fig. A). Based on the clinical features of the tongue lesion and on the patient’s medical background, the diagnostic hypothesis included metastatic melanoma, squamous cell carcinoma and fibrous hyperplasia.\nAn incisional biopsy was performed under local anesthesia. The histopathological analysis showed nests and solid sheets of non-pigmented atypical cells with an epithelioid phenotype infiltrating the sub-epithelial connective tissue. A high mitotic index was observed (Fig. B). Tumor cells displayed diffuse immustaining for S100 protein. Other melanocytic markers such as HMB-45 and Melan-A were negative. (Fig. C).\nAfter the diagnosis of a metastatic lesion on the tongue, the patient was referred back to the clinical oncologist who maintained her in a palliative protocol of chemotherapy with cisplatin, dacarbazine and vinblastine (CVD). Unfortunately, the patient died due to disease progression four and a half months after the diagnosis of tongue metastasis. |
Case 1 was a 63-year-old man who presented with a 4-month history of productive cough and started to deteriorate after having a fever for a week. The patient had an 8-year history of cigarette smoking, and his father had been diagnosed with lung cancer. Repeated antibiotic treatment had been used since he started coughing, which produced a small amount of yellow sputum, and on January 6, 2018, he was diagnosed with community-acquired pneumonia. Unfortunately, his condition did not improve, and he gradually developed persistent right chest pain and shortness of breath after activity. He was admitted to our hospital on March 19th, and chest computed tomography (CT) showed an irregular solid nodule in the anterior segment of the upper lobe of the right lung and lamellar density shadows in the lower lobe of the right lung ( and ). A bronchoscopic examination showed that the bronchi were unobstructed, slightly congested and had a mild amount of purulent exudate. A transbronchial lung biopsy (TBLB) was performed in the posterior basal segment and dorsal segment. Galactomannan (GM) of bronchoalveolar lavage fluid (BALF) was 1.068. No fungus, interstitial fibroblastic proliferation, incrassation or fibrosis was seen in the lung tissue biopsy, but acid-fast staining and PAS negativity suggested organizing pneumonia; the patient was then diagnosed with organizing pneumonia. The patient was started on prednisone 30 mg orally once a day on March 27, and he was discharged after his symptoms of cough, chest pain and shortness of breath had gradually improved. He took prednisone as prescribed after discharge. However, his condition worsened with a fever that developed on May 8, and he was admitted to the hospital on May 15. On examination, his body temperature was 39.6°C, and his superficial lymph nodes were not swollen. A small moist rale was detected in the lower right lung. Routine blood tests revealed a leukocyte count of 15.85×109/L, a red blood cell count of 4.18×1012/L, a hemoglobin level of 110.70 g/L, and a neutrophil percentage of 83.0%. His hypersensitive C-reactive protein was 171.23 mg/L, the erythrocyte sedimentation rate was 70 mm/h, and serum procalcitonin was 0.707 ng/mL. The serum was positive for AIGAs. His plasma HIV antibody, blood culture, beta-D-glucan, GM and Cryptococcus latex agglutination tests were all negative. The lesion in the right upper lobe was still present, and the high-density lesions in the posterior and outer basal segments of the lower lobe of the right lung were slightly smaller on May 16. A percutaneous lung biopsy was performed on May 21, 2018. The right lower lung biopsy showed a chronic inflammatory lesion but no granuloma or neoplastic lesion. Immunohistochemistry with PAS and acid-fast staining showed that the chronic inflammatory lesions were negative. After the admission of the patient and after considering an infection in the lung, the dosage of prednisone was gradually reduced and ultimately discontinued on May 24. The patient was successively treated with imipenem-cilastatin, voriconazole, compounded sulfamethoxazole and linezolid but still had a fever, with the highest temperature being 39.1°C. On May 30, his symptoms, including difficulty breathing, gradually worsened, and he developed type I respiratory failure. Therefore, he was transferred to the intensive care unit (ICU) for endotracheal intubation for salvage treatment. After admission to the ICU, the patient had scattered herpes in his right anterior chest wall and posterior chest wall. Herpes zoster was clinically diagnosed, and acyclovir sustained release tablets were given as oral antiviral treatment. T. marneffei was cultured from lung biopsy tissue performed on June 4, and chest CT showed growth of the two lung lesions ( and ). Liposomal amphotericin B was started on June 6 as an antifungal agent; the patient’s temperature returned to normal, and the patient’s cough and shortness of breath improved after 3 days. He was transferred back to the general ward. Unfortunately, liposomal amphotericin B was discontinued due to progressive renal impairment and was replaced by voriconazole 0.2 g twice daily starting on June 19. He was discharged after a chest CT showed that the lung lesions were significantly smaller ( and ) on June 25. He was started on a course of itraconazole after discharge. He developed another episode of cough and fever with a maximum temperature of 40°C on December 25, 2020. A chest CT showed that the nodules in the right upper lobe of the lung were significantly larger and that the lesions in the lower lobes of both lungs were stable on January 15, 2019 ( and ). Bronchoscopy revealed that the mucosa in the anterior segment of the right upper lobe had lost its normal structure, and the lumen was narrow. The pathology from the TBLB performed in the anterior segment of the right upper lobe showed primary adenocarcinoma (–). The patient was confirmed to have right lung adenocarcinoma and TSM. However, he was discharged after refusing treatment and died in February 2019. |
Our patient is an 89-year-old male with a history of a Hartmann’s procedure (2006) for Crohn’s disease of the sigmoid colon followed by a reversal of the colostomy with a diverting loop ileostomy (2008). Due to his comorbidities and age, the patient declined further surgery to reverse his loop ileostomy. Most recently, the patient presented to the emergency room with no ostomy output for the past 14 h, nausea and frequent burping. He reported a burning sensation around his ostomy. His physical examination revealed a large parastomal hernia in the right hemiabdomen and associated tenderness in the right upper quadrant, with no overlying skin changes. His white blood cell count was 8.1 (82.4% neutrophils), and he had normal liver function tests. A computed tomography (CT) scan of the abdomen and pelvis revealed a large parastomal hernia with a wide neck containing a distended gallbladder concerning for acute cholecystitis, as well as a small bowel obstruction with a transition point at the level of the hernia (Figs and ). A previous CT scan showed the same hernia with a nondistended gallbladder and non-obstructed bowel entering and exiting the stoma (Fig. ).\nThe patient was diagnosed with acute cholecystitis causing a small bowel obstruction within a parastomal hernia. A nasogastric tube was placed with return of 300 ml of bilious fluid. The patient was admitted and started on IV antibiotics for his acute cholecystitis. He had cardiopulmonary comorbidities and was deemed a poor surgical candidate by his cardiologist, so the patient declined any further surgeries. As his acute cholecystitis resolved with antibiotics, his ileostomy began functioning and the nasogastric tube was removed. His diet was advanced and his ostomy continued to function. His hospital course was complicated by a chronic obstructive pulmonary disease exacerbation which improved with steroids and nebulizer treatments. The patient recovered well and was discharged home on hospital day 5. |
A 53-year-old woman was admitted to our department with two episodes of painless gross hematuria during the last three months. She had no history of systemic diseases, constitutional symptoms and familial history of urological disorders. Physical exam revealed a palpable ill-defined mass in the right upper quadrant with extension to the umbilical region. Serum creatinine was 1.4 mg/dL, and urine analysis revealed microscopic hematuria (20-25 RBC / HPF). Urine cytology was negative for malignancy. Intravenous Urography (IVU) revealed left side fused crossed renal ectopia and filling defect within the pelvis of the crossed kidney (). A filling defect was evident in the left crossed fused kidney in retrograde pyelography (). Computerized-tomography (CT) scan confirmed the presence of left crossed renal ectopia associated with an enhancing mass, arising from the pelvis and pyelocaliceal system of the crossed kidney (, ). Cystoscopy showed no abnormality within the bladder. Ureteral orifices were normal. Chest X-ray and liver function tests were normal. The patient underwent surgery applying a midline incision. Left kidney showed a lump pattern, embedded in the lower pole of the right kidney. Both pelvises of kidneys were located anteriorly. Left renal pelvis and parapelvis tissue were firm.\nThe main renal arteries were dissected thoroughly. Interestingly, when the left main renal artery was clamped temporarily, both kidneys became partially ischemic, whereas clamping the right renal artery was associated with right renal ischemia with no effect on the crossed left sided kidney. During the right renal artery clamping, left nephrectomy was performed. All bleeding sites were ligated with absorbable sutures. Left ureter was resected near the bladder. Frozen section from margins indicated no evidence of tumor involvement. No palpable regional lymph node was detected. Post-operative course was uneventful. Macroscopic evaluation of the specimen revealed a 9 × 7 × 5.5 cm kidney with a well-defined mass, which had variegated cut surfaces measuring 8 × 5 × 4 cm with extension into the proximal ureter.\nMicroscopic evaluation showed a malignant renal tumor with solid and alveolar patterns, clear cell morphology and severe nuclear atypia (Fuhrman grade 4/4). Immunohistochemistry evaluation showed negative reaction for CK7, CK20, CK5 and CK6, and high nuclear weight cytokeratin in favor of clear cell type renal cell carcinoma.\nRenal sinus and pyelocaliceal system were invaded by tumors. However, the perirenal fat was tumor free. No vascular invasion was noted and all surgical margins were tumor free. Magnetic Resonance Imaging (MRI) and ultrasonography was applied on a regular basis to detect any evidence of local recurrence. Nevertheless, local recurrence was not detected during the 18-month follow up after surgery (). |
A 65-year-old man who was on maintenance HD for 27 years presented with gross hematuria for which subsequent imaging was highly suggestive of renal malignancy. He underwent left radical nephrectomy as it was a local disease and pathology confirmed RCC. Two years after the nephrectomy, he was unfortunately diagnosed with metastatic RCC. His past history was significant for Child-Pugh class B hepatitis C (HCV) related liver cirrhosis (albumin 28–35 g/L, Bilirubin <34 μmol/L, INR <1.7, presence of small amount of ascites, one past episode of grade 2 encephalopathy). The only episode of hepatic encephalopathy occurred 15 months before the diagnosis of metastatic RCC and was managed successfully with lactulose; he was subsequently started on rifaximin and had remained well since with no recurrence of encephalopathy. After deliberation of the various approaches for treatment, his oncologist considered TKI to be the best treatment option and started him on sunitinib 25 mg daily.\nOur patient was brought to the emergency department (ED) with confusion and bilateral asterixis 44 days after initiation of sunitinib (patient was maintained on 25 mg daily throughout this period). After appropriate investigations including computed tomography (CT) of the brain had ruled out other causes, he was managed as for metabolic encephalopathy (grade 2). His serum ammonia level was 170.5 μg/dL at presentation. His symptoms resolved 4 days later (with discontinuation of sunitinib). His serum ammonia was reduced to 48.7 μg/dL. He received the same dialysis schedule during his hospitalization and was discharged to continue with his usual HD regimen (3×/wk and 3.5 h/session).\nHe was later seen by his oncologist and was started at a lower dose of 12.5 mg sunitinib daily 2 weeks after his discharge, increasing to 25 mg daily 4 weeks after being stable on the lower dose with no complaints. He presented to ED again with similar presentation of confusion and asterixis 28 days later after the increase in dose. His serum ammonia level was significantly elevated again at 176.1 μg/dL pre-dialysis, declining to 68.2 μg/dL post-dialysis. He was dialyzed with a larger surface area high-flux filter (polysulfone 1.6 m2) using blood flow rate of 280 mL/min and dialysate flow rate of 500 mL/min for 4 hours. His body weight was 54.5 kg. His confusion improved significantly post dialysis and he recovered the following day. He was maintained on sunitinib 25 mg daily and discharged well with a schedule for more intensive dialysis (higher frequency 4×/wk and with better clearance achieved by increased blood flow, longer duration, and larger filter). He was successfully kept out of hospital with no recurrence of encephalopathy with that dialysis schedule. There were no adverse events and episodes of confusion.\nExcept for the elevation in serum ammonia level, there were no changes in coagulation profile, bilirubin, or liver enzymes during each of these 2 admissions for encephalopathy. There were also no other precipitating factors for hepatic encephalopathy that was evident in these admissions. He did not experience constipation and was compliant with lactulose and rifaximin. CT brain done during his admission did not reveal structural brain pathology. He does not consume alcohol and did not take traditional Chinese medicine. He was not given any medication that can inhibit CYP3A4. He was also not taking any therapeutic agent that has reported association with hyperammonemia. Patient was jointly managed by a multi-disciplinary team (Liver, Medical Oncology and Nephrology). All management decisions were discussed and made jointly by consensus. |
A 53-year-old male was brought to our Emergency Department with 1 month of confusion. He was completely normal 1 month prior, running a successful small business. The patient's wife initially observed confusion which worsened with impaired functioning, impairing his ability to work and care for his children. The patient was initially brought to an outside facility around 2 weeks into this illness. At that time, his symptoms had progressed to difficulty recognizing family, mutism, and hypersomnia. He then developed myoclonus with agitation and combativeness. MRI and CSF analysis from the outside facility were unremarkable (including an initially unavailable report of “unlikely prion disease” with no 14-3-3 protein detected). The patient was released with eventual admission to our hospital. By week 4 of his disease, he became bedridden, disoriented, and unable to recognize family. He developed myoclonic jerks and did not follow commands. Moreover, he had moderately increased muscle tone and incontinence. He never developed psychiatric manifestations. His clinical course continued to decline with subsequent intubation for airway protection by week 5 with deterioration by week 6 to GCS of 1/1/1. He has remained in this state ever since.\nPast medical history was significant for remote history of nasal carcinoma. The patient took no home medications. His surgical history was significant for nasal carcinoma resection. Family history consisted of a father with hypertension. The patient denied using tobacco or drugs and used alcohol socially only. He ran a busy automobile mechanic business. He traveled to El Salvador 1 year prior to presentation. There are no recent sick or animal contacts.\nAt our hospital, he was never febrile with stable vital signs. After his initial negative MRI at the outside facility, MRI head began to show bilateral caudate and basal ganglia abnormality on T2 as well as DWI. Additionally, there is a cortical ribbon involvement in the posterior portion of both hemispheres (Figure ,). There was never any pulvinar involvement. Routine complete blood count, complete metabolic panel, and coagulation studies were within normal limits. Serum analysis showed normal levels of vitamin B12, folate, methyl malonic acid, C-reactive protein, erythrocyte sedimentation rate, Cryptococcus antigen screen, Treponema antibody EIA, and ceruloplasmin. N-methyl D-aspartate (NMDA) receptor antibodies and antineuronal antibody panel were negative. HIV was non-reactive. EEGs showed periodic synchronized electric discharges which were refractory to antiepileptic drugs (Figure ). Repeat lumbar puncture was performed and showed WBC 86/mm3 with 38% neutrophils, 34% lymphocytes, and 8% bands, RBC 3048 mm3, glucose level of 104 mg/dL, and protein level 100 mg/dL. CSF herpes simplex virus PCR, VDRL syphilis screen, West Nile IgM and IgG ELISA, and AFB stain were all negative. Cytology showed inflammatory cells but no malignant cells. Repeat CSF 14-3-3 protein and RT-QuIC collected at 4 week after initial symptoms finally came back positive with probability of prion disease >98%. Brain biopsy, although requested, never took place.\nAfter 2 months of acute hospitalization but continued stability, patient was placed at a nearby subacute facility. Follow-up visits show no change nor deterioration. Patient remains on mechanical ventilation via tracheostomy and tube feeding at 3 year point from original symptoms. |
A 33-year-old Asian woman returned for a frozen embryo transfer (FET) cycle three years following an uncomplicated pregnancy and birth that resulted from a previous successful attempt at ovulation induction with gonadotropins and IVF. She had been placed on an antagonist protocol and her stimulation was unremarkable. Her medical history was significant for hyperlipidemia and oligoovulation secondary to PCOS, which was diagnosed at the time of her initial presentation. At that time, she had irregular menses occurring every 1–4 months and evidence of bilateral polycystic ovaries on ultrasound. Her androgen levels were normal and she did not exhibit any evidence of clinical hyperandrogenism. Prior to returning for FET, her menses remained irregular; however she denied the use of any hormonal agents. There was no personal or familial history of endometriosis or gynecologic malignancies. Pelvic ultrasounds (PUs) performed at her initial infertility consult as well as during treatment and in the early stages of pregnancy revealed polycystic-appearing ovaries, but were otherwise normal.\nPrior to the commencement of a FET cycle, the patient had a baseline PU, which revealed a complex ovarian cyst with layered low level echoes measuring 5.1 × 3.8 cm (). One area at the base of the cyst had a papillary growth (), while color Doppler revealed venous flow within the growth suggesting malignancy. Tumor markers including CA125 (21 U/mL) were within normal range. A repeat PU and a subsequent CT scan confirmed the previous ultrasound findings.\nAt exploratory laparotomy by gynecological oncologist, an enlarged fluctuant and cystic left ovary adherent to the rectosigmoid was found. The pelvis was otherwise normal. After obtaining peritoneal washings, the patient underwent a left salpingo-oophorectomy and omental biopsy but the cyst inadvertently ruptured during the procedure. Gross examination showed a 5 cm cyst with a 2.5 cm papillary lesion arising from the cyst wall. Final pathology revealed a stage 1c, grade 2 intracystic endometrioid adenocarcinoma arising in an endometriotic cyst and an ovary with changes including multiple cortical follicular cysts and stromal luteinization, consistent with polycystic ovarian disease. The omental biopsy and pelvic washings were negative for malignancy.\nThe patient's postoperative course was uneventful. Because there was spillage of the tumor during the initial procedure, the patient desired more aggressive management. She was reexplored and underwent a modified radical hysterectomy, right salpingo-oophorectomy, pelvic and paraaortic lymphadenectomy, appendectomy, and omentectomy. Final pathology revealed no residual disease. She received 6 cycles of cytotoxic chemotherapy with carboplatin and taxol. With regard to her reproductive plans, she is considering frozen embryo transfer with a gestational carrier. |
A 44-year-old Japanese man presented to the Maxillofacial Surgery Clinic at the Tokyo Medical and Dental University, Tokyo, Japan, with reports of a hard mass and dull pain in the left mandible. His medical history was generally unremarkable, though his mandible had been accidentally smashed against his young son’s head at the age of 40. No fracture was detected at the time, and no treatment was received.\nClinical examination revealed slight swelling in the left lower molar region and swelling of the left mandible. There was no disturbance of sensitivity in the left lower lip or chin, and he could move his lips normally. Radiographic examination showed large, irregular, multilocular radiolucency of the left area of mandible extending to the premolar and molar area, with no evidence of any impacted tooth (Fig. ). A computed tomography (CT) image revealed a 45 × 30 × 30 mm multilocular cystic mass in the mandible (Fig. ). An axial CT image showed thinning or partly resorption site of the buccal cortical plate. The coronal section images of the CT scan showed the mass lesion displacing the mandibular canal downward, near the inferior border. The roots of the adjacent teeth were resorbed, changing their shapes (Fig. ). The location of the teeth was not changed.\nThe initial clinical impression was an ameloblastoma, myxoma, keratocystic odontogenic tumor or another tumor type lesion.\nThe lesion was removed surgically with the buccal cortical bone and buccal mucosa in contact with the mass of the tumor. The mass fully excised intraorally under general anesthesia, and the inferior alveolar nerve in contact with the mass was preserved.\nThe resected specimen of the primary tumor was a soft, fragile, yellowish-white mass. Microscopically, tumor tissues were composed of plexiform or cord-like nests of tumor cells with round to oval nuclei and eosinophilic cytoplasm in a matrix with prominent myxoid change (Fig. ). On the other hand, the component of solid sheets of tumor cells was limited (Fig. ). In some areas, tumor cells surrounded small blood vessels (Fig. ). Moreover, tumor cells manifested a uniform cell morphology, poor dysplasia, and inconspicuous mitosis.\nImmunohistochemical staining was positive for vimentin (Fig. ), muscle-specific actin/HHF35 (Fig. ), and Calponin (Fig. ), a protein responsible for binding the actin-binding protein. A hairline-shaped area of positive staining for type IV collagen surrounding the tumor cells was also observed (Fig. ). VEGF and D2–40 was also focally positive in tumor cells. In contrast, staining for alpha-SMA, cytokeratin (AE1/AE3), cytokeratin (CAM5.2), CK19, CD34, CD68, p63, S-100, Factor VIII, and desmin, CD56, chromogranin A, synaptophysin was all negative in the tumor cells. Moreover, CD31 was also negative, but we observed many blood cells intervened between tumor nests. The Ki-67 labeling index was almost 1%. No necrosis or tumor invasion into the neurovascular channel was observed. The primary tumor was diagnosed as a glomus tumor based on these findings.\nThe patient remained symptom-free and manifested no signs of recurrence. However, a recurrent tumor was detected in a panoramic radiograph during an 8-year follow-up. The panoramic radiographs taken earlier, after excision of the primary tumor, showed normal healing process, bone regrowth, and increased radiopacity. The follow-up panoramic radiograph 8 years later depicted the recurrence as a radiolucent expansion in the lower area of the mandible. A CT exam showed an expanding lesion exiting the lower site of the mandible and a thinning buccal cortical bone in contact with the tumor (Fig. , ). In magnetic resonance imaging (MRI), the recurrent lesion showed a lower or compatible signal intensity compared to the muscle in T1-weighted images and a low-to-high inhomogeneous signal intensity in T2-weighted images. The apparent diffusion coefficient in the glomus tumor area was 2.0 × 10− 3 mm2/sec, suggesting a low cellular density (Fig. -).\nThe recurrent tumor mass and related teeth were removed under general anesthesia, and the bone surface was shaved. The recurrent tumor had the same microscopic, morphologic, and immunohistochemical features as the primary tumor (Fig. ). The tumor invaded the medullary cavity of the mandible, involving spongiosa and resorbing compressively the cortex bone around tumor.\nThe patient has no symptoms of recurrence as of this writing, 2 years after the second operation, and will be followed for the long term to promptly detect any signs of new tumor growth.\nThe patient is satisfied that the glomus tumor was regulated without resecting the mandibular region. He is pleased that there was no functional deterioration that he was worried about before treatment and there was no aesthetic damage.\nTimeline of patient diagnosis and treatment.\n1. 4 years before the first visit, the patient had been smashed against his young son’s head.\n2. At first visit, the symptom was slight swelling of the left mandible. There was no disturbance of sensitivity. The examinations were performed with X-lay and CT.\n3. First surgery was performed and started clinical follow-up after the surgery.\n4. 8 years later from first surgery, the recurrence was found by panoramic radiograph. The examinations were performed with CT and MR.\n5. Second surgery was performed, and it was passed 2 years with no recurrence from second surgery. |
A 51-year-old female with a 25 pack year smoking history and newly diagnosed right breast cancer underwent right skin sparing mastectomy with sentinel lymph node biopsy, left prophylactic skin sparing mastectomy and immediate tissue expander reconstruction at our institution. The patient had right breast stage IA multifocal invasive ductal carcinoma and multifocal ductal carcinoma in situ (DCIS). Margins were negative with invasive carcinoma closest at 0.7 cm and DCIS closest at 0.5 cm. Sentinel lymph node biopsy was negative for metastatic carcinoma. The patient denied history of coronary artery disease, myocardial infarction, congestive heart failure, diabetes mellitus, kidney or lung disease, venous thromboembolism, or any bleeding disorders. She likewise denied drug use or daily alcohol consumption. Having stopped smoking for several months prior to the surgery, the patient did not consider herself a smoker and reported herself as a “non-smoker”. On physical examination, she had 40 DD cup breasts with grade 3 ptosis. The patient was deemed an American Society of Anesthesiologists Physical Status of 2, indicating mild systemic disease. She was a good candidate for skin sparing mastectomy because her breasts were not pendulous. 700 mL fill tissue expanders were placed sub-pectorally with Flex HD support for the inferolateral breast pocket. Each expander was filled with 500 mL intraoperatively. At conclusion of the mastectomy, her flap appeared viable with good capillary refill. At completion of expander placement, her flaps continued to appear very viable. Her postoperative course was complicated by extensive bilateral mastectomy skin flap necrosis (). This was managed with tissue expander removal, debridement and primary closure two weeks after her initial surgery given the need to start chemotherapy. It was only after post-hoc questioning that the patient revealed that she was actively engaging in e-cigarette use. Indeed, she noted switching to e-cigarettes approximately 3 months prior to her surgery due to the belief they were safer than traditional cigarettes and maintained a usage similar to her previous 1.5 pack per day amount. |
A 37-year-old asymptomatic female patient was referred to our hospital with an enlarged cardiac silhouette found on her screening chest X-ray (Fig. , panel ). She had no history of cardiovascular disease. She had a spontaneous abortion 3 years previously, and she is a mother of three children. She has a positive family history of cardiovascular diseases and cancer (lung adenocarcinoma and brain tumour). Physical examination revealed distant heart sounds, a regular rate and rhythm. The echocardiographic examination revealed pericardial effusion and an inhomogeneous mobile mass located in the pericardial sac around the left ventricle (Fig. , Additional file ). Subsequently, cardiac magnetic resonance imaging (MRI) was performed, which showed an intrapericardial, semilunar-shaped mass with a size of 10 × 4 × 9 cm attached to the pulmonary trunk surrounding the aortic root, left atrium and left ventricle (Fig. ). The tumour had well-demarcated margins and did not invade the blood vessels or myocardium. The caudal part of the tumour was mobile, while its cranial part was fixed to the pulmonary trunk (Additional file ). The MRI scan detected intermediate signal intensity on proton density-weighted images and high signal intensity on T2-weighted spectral fat saturation inversion recovery (SPIR) images (Fig. , panel ). Both first-pass perfusion and early and late gadolinium-enhanced (LGE) images showed that the mass was vascularized and showed septated, patchy, inhomogeneous LGE (Fig. , panel ). Coronary computed tomography angiography (CTA) was performed to see whether the coronary arteries were affected. The coronary CTA proved that coronaries were not invaded by the tumour (Fig. ). We have evaluated the previous screening chest X-rays of the patient that were acquired during the past 10 years. Based on these chest X-rays, we can conclude that the mass had started to form at least 7 years ago (Fig. ). No abnormalities were found by the abdominal ultrasound examination, the results of hormone tests were normal, and a hormone-secreting nature of the tumour was excluded. Open heart surgery was indicated through median sternotomy (Fig. , panel ). The intraoperative findings confirmed the MRI and coronary CTA results. The tumour was intrapericardial, attached to the lateral wall of the pulmonary trunk, 2 cm distal from the commissures of the pulmonary valve. The tumour did not invade any other structures of the heart. Complete resection of the tumour with partial resection of the pulmonary trunk was performed (Fig. , panel ) using cardiopulmonary bypass. The pulmonary trunk was reconstructed with a round-shaped bovine pericardial patch (Fig. , panel ). The intra- and postoperative course were uneventful. Histological evaluation of the septated, cystic mass revealed tumour cells forming an irregular pattern, the so-called “patternless pattern” (Fig. , panel ). Immunohistochemically, the cells tested positive for vimentin, CD34, CD99 and STAT6 (Fig. , panel ) but negative for keratin (AE1-AE3), CD31 and S100. Thus, the diagnosis of a primary cardiac solitary fibrous tumour (SFT) was established. The tumour was classified as non-malignant because of the lack of increased mitotic activity, an intact capsule and no sign of vascular invasion. Regular and long-term clinical and MRI follow-up were indicated (every 6 months in the first year, later annually) because of the risk of late local recurrence. At the 3-year follow-up, the patient had no symptoms, and MRI did not show recurrence of the tumour (Fig. ). The patient’s clinical history is summarized in a timeline, prepared in accordance with CARE guideline (Additional file ). |
A 63-year-old Japanese woman was referred to our department because of an abnormal shadow at the left side of her chest wall on computed tomography. She had undergone total hysterectomy and radiotherapy for cervical carcinoma 4 years prior. One year after the first surgery, three metastatic lung nodules appeared at the upper lobe of her right lung, the lower lobe of her right lung, and the lower lobe of her left lung. Wedge resection for upper and lower lobe of her right lung was initially performed via three-port thoracoscopic surgery. Then, wedge resection for the lower lobe of her left lung was performed via eighth intercostal single incisional thoracoscopic surgery. After the surgery, an intrathoracic chest wall mass developed which increased in size gradually. Her gynecologist introduced her to our department for surgical resection of the mass. Her family, including her parents and two sisters, had been healthy and had no inheritable diseases. She had no symptom, drug history, tobacco smoking history, or psychosocial history, and she was a social drinker. She had not received any medications since the mass developed and until admission to our hospital. She had undergone an operation three times as mentioned above and had been a carrier of type B hepatitis.\nAfter her admission to our department, her general condition was good, and there were three operative scars at both sides of her chest and lower abdomen. Her chest sounds were clear and there was no neurological abnormality. She was 151.1 centimeters tall and weighed 49.8 kilograms. Her heart rate was 77/minute, blood pressure was 135/87 mmHg, and body temperature was 36.1 °C. The laboratory findings were white blood cells 5.25 × 103/μL, hemoglobin 12.7 g/dL, and platelets 156 × 103/μL. A liver function test revealed: albumin 4.6 g/dL, aspartate aminotransferase 15 U/L, alanine aminotransferase 13 U/L, and total bilirubin 0.3 mg/dL. A renal function test revealed blood urea nitrogen 13.6 mg/dL and creatinine 0.79 mg/dL. An electrolyte test revealed sodium 143 mEq/L, potassium 3.8 mEq/L, and chlorine 106 mEq/L. A tumor marker test revealed carcinoembryonic antigen 3.4 ng/mL and squamous cell carcinoma antigen 0.80 ng/mL. Another test revealed positive reaction to type B hepatitis surface antigen and C-reactive protein < 0.1 mg/dL. Computed tomography demonstrated a gradually increasing low-density mass measuring 2.0 × 1.8 cm in diameter (Fig. ). Magnetic resonance imaging demonstrated a low-intensity mass in T1-weighted imaging and a high-intensity mass in T2-weighted imaging (Fig. ). The mass was thought to be a singular cyst; however, this type of cyst was rare and the mass was increasing. Therefore, dissemination of cervical carcinoma could not be excluded, and surgical removal of a part or tissue of the mass was performed.\nIn the right lateral position, thoracoscopic excision of the mass was done with two ports (3 mm and 2 cm access ports) by two general thoracic surgeons (Fig. ). First the 3 mm port was set at the sixth intercostal space on the inframammary line. Most of her left lung was attached to her chest wall; therefore, the second port was set above the cyst and lysis of adhesions was done. After the lysis, the cystic mass was found adhering to the upper lobe of her left lung. The adhesion of the mass to her lung was not strong and could be separated without injury to the visceral pleura. Therefore, the mass was thought to derive from the chest wall pleura and was resected by adhesiolysis.\nThe mass was a unilocular cyst containing mucinous fluid. On microscopic examination, the cyst was lined with a single layer of cuboidal epithelium (Fig. ); immunohistochemistry showed positive staining of calretinin and D2-40 (Fig. ). Thus, the cyst was diagnosed as mesothelial cyst derived from the chest wall pleura. Five years after the surgery, our patient had no evidence of cyst or cervical carcinoma on computed tomography. |
During routine dissection of a 69-year-old fresh female cadaver at the Department of Anatomy at our institution, variations in the arterial system of both upper limbs were noted. There was no history or evidence of any invasive procedure in the upper limbs of that person.\nOn both sides, the brachial artery in the middle third of the arm originated a SuBUA (Figures and ). This artery penetrated the brachial fascia in the lower third of the arm, crossed anteriorly to the bicipital aponeurosis and to the muscles that originated from the medial epicondyle, and ran over the pronator teres muscle in a doubling of the antebrachial fascia (Figure ). In the elbow region, the SuBUA was in intimate contact with the superficial structures, namely the medial antebrachial nerve and the subcutaneous veins (Figure ). It then dipped into the forearm fascia, passed through a gap between the palmaris longus and the flexor carpi radialis, ran deep to the palmaris longus muscle belly, and superficially to the flexor digitorum superficialis muscle, reaching the gap between the latter and flexor carpi ulnaris muscle (Figure ). In the middle third of the forearm the SuBUA was positioned lateral to the ulnar nerve.\nThe brachial artery continued through the radial artery (RA), which followed its usual course. In the upper third of the forearm, the RA gave off the common interosseous artery. This latter artery branched into the anterior and the posterior interosseous arteries (Figure ). The anterior interosseous artery had a large caliber and originated branches to most of the anterior compartment muscles. The radial recurrent artery emanated from the radial artery, and the anterior ulnar recurrent artery was a branch of the common interosseous trunk.\nIn the distal third of the forearm and in the wrist region, the RA and the SuBUA divided in the same manner as the radial and ulnar arteries usually distribute [], originating the superficial and deep palmar arterial arches. Figure schematically portrays the distribution of the RA and the SuBUA in the cadaver herein described. |
A 10-year-old boy, practicing karate since the age of six, presented with acute pain in his left wrist after falling on the outstretched hand. On physical examination, there was tenderness in the anatomical snuffbox and a painful range of motion of the wrist. Plain radiograph showed a fracture of the scaphoid waist with sclerosis, central cystic bone alteration, and deformity of the proximal pole suggesting an ongoing AVN (). Additional CT scan supported the radiographic findings with a more pronounced central cystic bone alteration and irregularity of the fracture border (). The diagnosis of AVN was confirmed with signal loss of the scaphoid on MRI T1 (). A dynamic contrast-enhanced MRI was performed for further assessment of the proximal pole vascularity and treatment planning. Therefore, a region of interest was placed on the proximal and distal scaphoid poles. Time-signal intensity curves were recorded and were considered to represent the degree of vascularity. The time-signal intensity curves are classified into good, fair, or poor vascularity based on their shape and maximal enhancement comparing proximal pole with the distal pole [, ]. In this study, the time-signal intensity curve on the proximal pole was lower than the distal pole with a maximum enhancement of 50%, defining fair perfusion of the proximal pole. The fair perfusion of the proximal pole on dynamic contrast-enhanced MRI was with the account of measuring associated fibroblasts in the cystic alteration in the distal pole (). Because of fair perfusion of the proximal pole, an adequate healing potential with conservative treatment was estimated. The wrist was immobilized with a short arm cast for fourteen weeks and at the end of cast immobilization the patient was pain-free and had no tenderness and there was no restriction in range of motion. Plain radiograph showed improved consolidation and CT scan performed one month later confirmed union of the fracture (Figures and ). |
A 35-year-old primipara with a dichorionic twin pregnancy came for a scheduled scan at 27 weeks. A minor hyperechoic structure located superior and anterior of the cerebellum was observed in twin 1 (). At a previous scan at 18 weeks no deviant findings were noticed. TORCH-serology was negative. At 31 weeks the biparietal diameter (BPD) was substantially enlarged, and so was the occipito-frontal diameter. A homogenous ovoid cyst-like structure measuring 65 × 53 mm and containing concentric rings was seen in the posterior part of the skull. The ventricular system was clearly widened. Doppler examinations, including the fetal middle cerebral artery, were normal and remained so throughout pregnancy. No turbulent flow was observed at any occasion within the cystic structure or elsewhere (). Tests for idiopathic thrombocytopenia and thrombocyte antibodies were negative. At 32 weeks BPD, although still enlarged, had decreased as well as the cyst-like structure (). The next scan, at 33 weeks, showed a continuous regress (). Days later, still in week 33, an MR examination showed a large expansive lesion in the back of the skull compressing cerebellum. At the bottom of the lesion a rounded structure was seen, interpreted as a thrombus or hemorrhage. The fourth ventricle was compressed leading to obstructive hydrocephalus. The parenchyma was assessed as normal (). The last scan was done at 35 weeks and showed a substantial regress of the cystic structure. BPD was still enlarged and the hyperechoic structure still visible (). Delivery was now undertaken through a caesarian section. Twin 1, a boy, weighed 2232 g and had an Apgar of 8 at 1 min and 10 at 5 min.\nTwin 1 was examined by ultrasound, MRI and computed tomography a few hours after birth and a partly thrombosed DSM was diagnosed. Later, MR examinations have exhibited a normalization including recanalization of vessels. The child received no specific treatment.\nTwin 1, now more than three years old, has up to now displayed normal growth and neurological development, well in line with his twin brother. |
A 36-year-old male patient was taken to our hospital by his family members because of elevated mood and aggressive behaviors. His medical history was collected by detailed inquiry. In his early adolescence, the patient experienced his first episode of depression, which was ascribed to the bad relationship between his parents. Since then, he complained that his mood had swung frequently, with declined interest in daily activities and loss of appetite. This patient condemned himself as solitary, endocentric, and cowardly. He had been a Christian for approximately 10 years. After graduating from university, he worked as a physics teacher in a local high school. His job performance at school was proficient. Not until this patient heard of health education on mental illnesses did he first come to a local clinic for psychiatric counseling (nearly 8 months prior to admission). On that occasion, he complained of feeling upset, irritable, and fatigued. He was diagnosed with generalized anxiety disorder and was prescribed tandospirone and propranolol. Nonetheless, these regimens were discontinued by the patient after 2 weeks because of the unsatisfactory outcome. He then visited our hospital and the psychiatrist revised the diagnosis to major depressive disorder. He was given sertraline at a dose of 100 mg/day. About 2 months after initiating sertraline treatment, this patient still felt depressed and was not interested in daily activities. The dose of sertraline was, therefore, increased into 150 mg/day. From then on, our patient had felt at ease with sertraline treatment for nearly 4 months. His performance at work and interpersonal relationships also improved.\nAbout 1 week prior to admission, the patient revisited our hospital and complained of waking up early in the morning, however his emotional state was stable. Trazodone at a dose of 25 mg/night was added to promote sleep. He felt extremely excited after taking trazodone for the first time; he slept only 2 hours that night, and became talkative and conceited. In the following 2 days, he continued to take trazodone, however on day 3 he discontinued trazodone due to abnormal behavior. He preached Christianity during his class which was forbidden according to the school disciplines. He even sent emails to the White House to seek protection. Also, our patient professed that he was conferred superpowers by God. The day before admission, the patient felt that he was possessed by a supernatural spirit and was told to save the world before the coming doomsday. He abused his superiors at a public occasion because of their insincerity. Consequently, he was sent to our hospital for management.\nOn admission, the patient could not stop talking and asserted the ability to understand the inner world of others. An extensive physical examination revealed no neurological deficits. No substance abuse or toxin exposure was reported. Laboratory screenings, including routine analysis of blood, urine, stool, biochemistry, thyroid hormones, coagulation function, C-reactive protein, cortisol, adrenocorticotropic hormone, tumor and infectious biomarkers, were all within the normal limits. Cranial magnetic resonance scanning was also normal. The patient was diagnosed with bipolar I disorder, manic episode, according to the Diagnostic and Statistical Manual of Mental Disorders, 5th edition. Quetiapine was gradually titrated to 700 mg per night over 1 week accompanied with 5 mg of haloperidol twice a day for 5 consecutive days. His emotional state gradually stabilized after about 2 weeks of hospitalization. After discharging from hospital, the patient took quetiapine 700 mg every night and remained euthymic during outpatient follow-ups. |
A 38-year-old male complaining of recurrent episodes of melena for the past 2 weeks was admitted to the emergency department of our hospital. Two days before the hospital visit, the patient had complained of the presence of blood in vomiting and had a history of recurrent complaints of upper abdominal pain often radiating to the back. His previous medical history revealed that he had suffered from chronic pancreatitis for 3 years before present admission, possibly due to alcohol abuse. He was resuscitated with the blood and intravenous fluid for melena during present admission. Ultrasonography (USG) whole abdomen revealed a moderately enlarged liver (18.5 cm × 12.3 cm) with heterogeneous echotexture, with a prominent portal vein, of size 1.3 cm; hepatic and splenic hilar collaterals were suggestive of portal hypertension. The presence of mild ascites was documented. Transabdominal USG showed echogenic pancreas with multiple, small, hyperechoic, nonshadowing foci in the pancreas suggestive of fibrotic changes. There was evidence of irregular dilated main pancreatic duct with pancreatic and intraductal calculi. These features were consistent with chronic pancreatitis. The findings were suggestive of alcoholic liver disease with portal hypertension and chronic obstructive pancreatitis. After resuscitation, upper GI endoscopy was performed on two different occasions that showed bleeding from the second part of the duodenum with an abnormal tortuous vessel at the ampulla of Vater. This was diagnosed as hemosuccus pancreaticus, which might have caused the bleeding []. Serendipitously during the endoscopy, a worm was also found penetrating the mucosa of the duodenum. The worm was retrieved and referred to the laboratory for identification. It was a single intact live worm (size 12.3 mm × 0.3 mm) reddish pink, with head bent in the same direction of the body curvature with a spine at the posterior end []. The buccal capsule had six teeth (four hook-like on ventral surface and two knob-like on dorsal surface) []. The live worm was seen to lay eggs while examining under the microscope []. Three consecutive stool samples of the patients were examined. The stool sample was dark-colored, semi-solid in consistency without any parasitic elements. Wet mount examination of stool revealed nonbile-stained ova (60– 65 μm in length, 40– 45 μm in width) with transparent hyaline shell membrane containing blastomeres. The above morphological features of the worm and micrometry of the ova were consistent with Ancylostoma duodenale. For further confirmation and to exclude the probability of mixed infection with Strongyloides stercoralis, the modified Harada– Mori nematode larval culture method[] was performed, and larva of hookworm was retrieved after 1 week of incubation at ambient room temperature. However, duodenal biopsy could not be done to rule out this possibility.\nThe patient was a chronic alcoholic and had deranged liver functions. Other laboratory test revealed low hemoglobin (5.6 g/dL), elevated total leukocyte count (12,300/μl) with increased eosinophils (neutrophils - 54%, lymphocytes – 24%, eosinophils – 16%, and monocytes – 6%), hyperbilirubinemia (total serum bilirubin 12.5 mg% and conjugated bilirubin 11.7 mg%), alanine transaminase/aspartate transaminase (87.7/90 IU), serum alkaline phosphatase (827 IU/L), hypoalbuminemia (2.3 g%), and raised serum globulin (3.9 g%) with reversal of albumin:globulin ratio. Blood and urine cultures were bacteriologically sterile. Peripheral blood smear examination showed microcytic, hypochromic anemia, with the absence of any hemoparasites. The patient was planned for contrast-enhanced computed tomography (CECT) abdomen, but the course of illness was rapidly fatal, and he succumbed to the illness within 48 h of admission, hindering further investigative course. The cause of death was unexplained shock secondary to upper GI bleeding precipitated by chronic, alcoholic liver disease with chronic pancreatitis and portal hypertension. |
A 3-year-old girl was admitted to our department with complaints of back pain since 1 year and progressive difficulty in walking since 6 months. Her mother declared she had been aware of spasticity in lower extremities since 6 months and for the last few weeks, she was unable to walk independently and had urinary incontinence. Her neurological examination revealed spastic paraparesis and bilateral extensor Babinski signs and bilateral clonus of the ankles. Her magnetic resonance imaging (MRI) revealed T8-9 intramedullary mass showing increased signal intensity in both T1- and T2-weighted images []. The patient was operated with a T8-9 laminoplasty and debulking and internal decompression of the tumor was made. Complete resection was not attempted as there was no plane of cleavage between the cord and the tumor. The dura was easily closed primarily without using a graft. Histologically, the tumor was uniformly composed of mature adipose tissue revealing a lipoma. Postoperative period was uneventful and neurological status of the patient get better immediately following surgery. She was mobilized with aid on the postoperative 3rd day and at the 1st month follow-up visit she was walking independently with a sequela of right ankle spasticity. First month control MRI revealed the rest mass almost the half volume of the preoperative mass. No strict dietary measures were taken but her mother was told to be careful about the weight gain of the patient as this might have the potential of increasing the tumor size. The 1st year MRI was almost the same and neurological status was same. The 2nd year MRI revealed the size of the lipoma was spontaneously decreased to almost half of postoperative size and 3rd year was the same as the 2nd year with a mild kyphosis []. The patients were observed to gain weight at the lower limit compared to charts of healthy children at this 3 years period. |
A 72-year-old woman with liver cirrhosis confirmed based on hepatitis C virus serology presented with profuse melena. Initial abdominal computed tomography showed paraesophageal varices and venous collaterals around the duodenum. Extravasated contrast was identified in the second portion of the duodenum with the afferent collateral vessel originating directly from the main portal vein. The efferent collateral vessel drained into the inferior vena cava via the ovarian veins (Fig. ). Emergent upper endoscopy was immediately performed, and bleeding duodenal varices in the second portion of duodenum were identified (Fig. ). Endoscopic band ligation was attempted first, and B-RTO was prepared simultaneously. Unfortunately, B-RTO following EBL failed because of unsuccessful cannulation of the ovarian vein, and there was a surgical treatment request. Laparotomy was performed under general anesthesia, and the venous collaterals around the second portion of the duodenum were identified and cannulated using an 18-gauge needle (Fig. ). Following cannulation of the venous collaterals, intraoperative angiography revealed portal vein and paraesophageal varices (Fig. ). The efferent venous collateral was ligated to the side of the needle entry point, and intraoperative angiography revealed duodenal varices, portal vein, and paraesophageal varices (Fig. ). The amount of contrast injected into the portal vein during intraoperative angiography was estimated. The afferent venous collateral was then ligated centrally over the needle entry point, and intraoperative angiography identified the ovarian veins and inferior vena cava (Fig. ). The remaining venous collateral was then ligated to the side of the needle entry point, and 6 mL ethanolamine oleate (EO; Grelan, Tokyo, Japan) solution with iopamidol 300 (5 % EOI; Schering, Berlin, Germany), which was the amount of contrast injected into the portal vein during the previous intraoperative angiography, was slowly injected into the afferent collateral vessel under intraoperative angiography, completing the operation. Endoscopic examination on postoperative day 4 showed embolization of the duodenal varices (Fig. ). The patient was discharged on postoperative day 11; however, she died because of liver failure 3 months after this operation without recurrence of the melena. |
A 48-year-old healthy, athletic Japanese female with a past medical history of right renal agenesis presented to the emergency department after being found unconscious by her husband. This occurred just minutes after she reported having severe 10/10 chest pressure with radiation to her jaw. Her husband called 911, and emergency medical service arrived within 7 min. She was found to be in ventricular fibrillation arrest and CPR was immediately started. No epinephrine was administered, but she was defibrillated twice with return of spontaneous circulation. Subsequently, she was brought to the hospital for further evaluation.\nThe patient states that she has never had this before and has no prior cardiac history. A detailed review of systems included 2-3 loose stools a day, for several days, which started shortly after completing a 30-mile marathon earlier in the week. She reports being unable to finish the run, due to recurrent anginal symptoms. Otherwise, she denied any significant symptoms. Her family history consists of a mother with breast cancer and a father with no significant medical history. Socially, the patient is married and just moved to the United States from Japan 2 years ago. She quit smoking 15 years ago and previously was smoking 6 cigarettes a day for approximately 10 years. She denied any prior recreational drug use. She reports drinking one beer a day.\nHer emergency room labs included a normal troponin, as well as hypokalemia at 2.7. Her ECG showed normal sinus rhythm, no ST segment elevations or depressions, and nonspecific T wave abnormalities (). She was started on medical therapy with aspirin 325 mg po daily, 80 mEq of potassium, metoprolol 12.5 mg BID, and a heparin drip. Her follow-up studies demonstrated minimally uptrending troponins after 6 hours, with a peak level of 0.18 ng/ml. A transthoracic echocardiogram was completed and was unremarkable, demonstrating preserved left ventricular systolic function and normal wall motion.\nA subsequent cardiac catheterization was performed, which showed no obstructive atherosclerotic disease. She did have mild mid left anterior descending artery stenosis, which did improve with intracoronary nitroglycerin (). Otherwise, her coronary arteries were normal.\nHer clinical course within the first 24 hours of admission was complicated by recurrent episodes of chest pain, associated with dynamic ST elevation on ECG (). She subsequently developed nonsustained ventricular tachycardia (Figures and ). Metoprolol was discontinued, and she was started on amlodipine 5 mg and isosorbide mononitrate 30 mg daily. An external defibrillator LifeVest was ordered.\nAdditional work-up at this point included a procainamide drug challenge, in order to exclude Brugada syndrome. The patient was given 10 mg/kg of procainamide over 10 min. Her baseline ECG demonstrated normal sinus rhythm, with no notable ST segment abnormalities in the precordial leads V1-V2. No changes in the ST segments or T waves occurred with the procainamide drug challenge. Other possible etiologies for recurrent ventricular arrhythmias were considered but were felt to be less likely the cause in her case: long QT interval (normal QTc on exam and all other ECGs), catecholaminergic polymorphic ventricular tachycardia ((CPVT) VT events were monomorphic and occurred at rest), short QT, and early repolarization (no ECG evidence). Autoimmune etiology was excluded with negative serology including antinuclear antibody, anti-double-stranded deoxyribonucleic acid, and rheumatoid factor. A cardiac magnetic resonance imaging was scheduled to evaluate for infiltrative disease and arrhythmogenic right ventricular cardiomyopathy (ARVC).\nGiven the presentation of cardiac arrest and her ongoing nonsustained ventricular tachycardia, the tentative plan at this time is to proceed with ICD implantation. After the initiation of the calcium channel blocker and long-acting nitrate, the patient's symptoms and events completely subsided. She was discharged home on these medications and the LifeVest, with a follow-up appointment for ICD placement and magnetic resonance imaging. A couple of weeks after, her MRI was negative for any infiltrative disease and ICD was implanted. |
A 75-year-old man was admitted to Inha University Hospital with a three-month history of language deterioration. He was also presenting gait disturbance and urinary and fecal incontinence. He had an eight-year history of hypertension and had undergone stent insertion for myocardial infarction five years previously. In computed tomography (CT) and magnetic resonance imaging, a 6.5 cm-sized heterogeneous enhancing mass with central necrosis was seen in both the frontal lobes and the anterior genu of the corpus callosum. Radiologically, the mass was suspected to be lymphoma or glioblastoma multiforme, and a stereotactic biopsy was therefore performed. A piece of the biopsy sample was sent for frozen section evaluation. The cytologic crush material was composed mainly of large single cells with indistinct cytoplasmic borders in a granular background. The tumor cells had abundant eosinophilic granular cytoplasm and round oval nuclei with prominent nucleoli. Some tumor cells with mild nuclear atypia were also discerned (). No endothelial proliferation or mitosis was identified.\nThe biopsy samples for permanent section consisted of three pieces of brain tissue. They showed cellular lesions predominantly composed of infiltrates of large round cells without endothelial proliferation. Reticulin deposition was seen only in the endothelial cells. The tumor cells had abundant intracytoplasmic eosinophilic granules, either dispersed uniformly or confined to the cell periphery (targetoid appearance) (). The granules were positive for periodic acid-Schiff (PAS) and resistant to diastase digestion. The nuclei were bland, round to oval, varied in size and often eccentrically located. Occasionally, mild atypical nuclei and small nucleoli were observed. In the entire field, no mitosis or necrosis was identified.\nThe granular cells were diffusely and strongly immunoreactive with GFAP showing cytoplasmic staining and targetoid appearance. The S-100 protein showed diffuse strong cytoplasmic immunoreactvity (). The tumor cells overexpressed the p53 protein and showed positive immunoreactivity for CD68 (KP), but were negative for CD68 (PGM). Epithelial membrane antigen (EMA), lysozyme, CD43, and albumin were not expressed. The Ki-67 labeling index was 5%. The patient was discharged without additional management. |
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