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A 39 year old male with a history of dog bite to the left lateral thigh during a police pursuit, was bought into the emergency room from jail complaining of acute onset vision loss in the right eye, shortness of breath, and left leg pain six weeks after the dog bite.\nThe wound was initially treated with oral antibiotics, and mildly improved. However, it became indurated later with painful tracking to the left hip.\nSocial history was significant for amphetamine abuse in the past.\nUpon presentation to the Emergency room, the patient was found to have tachycardia, and leukocytosis. The vital signs are summarized in table . The laboratory workup is summarized in table .\nFurther workup revealed left thigh abscess and signs of sepsis. The patient was transferred to the intensive care unit. Blood cultures showed oxacillin resistant staphylococcus aureus (ORSA) bacteremia, and further evaluation revealed multiple septic emboli to various organs, causing bilateral loculated pleural effusion (Figure ), left renal cyst abscess, left obturator abscess (Figure ).\nThe patient was initiated on intravenous levofloxacin, vancomycin, and gentamycin. He underwent emergent hip incision and drainage, computed tomography guided placement of bilateral pigtail tubes to drain the pleural fluid, and a nephrostomy tube was placed to drain the left renal cystic abscess. Trans-esophageal echocardiogram showed no evidence of endocarditis. The left hip arthrocentesis and the left renal cystic fluid both grew ORSA.\nMeanwhile, the ophthalmology service was consulted to evaluate the right eye acute vision loss in the setting of systemic ORSA bacteremia. Initial ophthalmological exam of the right eye showed visual acuity of 20/200 on the right and 20/25 on the left. Intraocular pressure was 10 mmHg bilaterally. Slit lamp exam of the right eye showed conjunctival injection, corneal edema, chemosis, anterior chamber cell and flare. Slit lamp exam on the left was normal. Dilated fundus exam of the right eye showed absent red fundus reflex, vitreous cells and debris, chorioretinitis and vitritis. Dilated fundus exam of the left eye showed two tiny cotton wool spots along the superior and inferior arcades.\nThe patient was diagnosed with endogenous endophthalmitis of the right eye as a complication of the systemic ORSA bacteremia, for which he underwent an anterior chamber tap and intravitreal injection of vancomycin, ceftazidime, and voriconazole in the right eye. Prednisolone (1 drop every three hours), and ofloxacin (one drop four times daily) were initiated.\nAntibiotics were continued including intravenous vancomycin, gentamycin, levofloxacin, and topical ofloxacin drops. The patient continued to be afebrile and white blood cell count decreased from 18.6 to 7.4.\nLater, the anterior chamber fluid aerobic, anaerobic, and fungal cultures came back all negative.\nFive days post injection exam showed visual acuity of 20/200 on the right, and 20/25 on the left. Intraocular pressure was 8 mmHg on the right, and 10 mmHg on the left. Dilated fundus exam showed decreased chorioretinitis and vitritis in the right eye. However the patient was noted to have macula-off rhegmatogenous retinal detachment, and a retinal hole inferior to the inferotemporal arcade (Figure , , ). Dilated fundus exam of the left eye showed decrease in size of the cotton wool spots on the superior and inferior arcades.\nThe patient was taken to the operating room to repair the retinal detachment. Encircling scleral buckling was performed. Inspection of the posterior pole revealed a macula-off rhegmatogenous detachment and a necrotic retinal hole inferior to the inferotemporal arcade. A posterior vitreous detachment was created by using the vitrector. Following this, the cryoretinopexy was applied to the superotemporal retina where there were some small atrophic holes near the buckle. The peripheral vitreous was trimmed using a scleral depression for 360 degrees without complications. Next, perfluorocarbon was placed on the macula to stabilize the macula, followed by endolaser photocoagulation to surround the retinal hole and also 360 degrees on the scleral buckle. Next, an air-fluid exchange was performed, followed by silicone oil placement. At the end of the operation there was good red reflex and the eye was normotensive by finger palpation.\nThe patient was continued on intravenous vancomycin and oral levofloxacin for systemic infection control, and was started on one drop of Maxitrol three times daily. Patient was advised to keep his head in a face-down position.\nPostoperative exam of the right eye showed flat retina 360 degrees (figure , ). The patient was discharged to jail with a plan to continue on intravenous vancomycin, oral levofloxacin, Maxitrol drops, Ofloxacin ophthalmic solution, Prednisolone eye drop, Atropine sulfate ophthalmic solution 1% four times daily, and Cosopt one drop twice daily.\nFollow-up exam 4 weeks post operation showed a visual acuity of 20/400 on the right and 20/25 on the left. Slit lamp exam of the right showed deep heme, 1+ cell, no signs of iris neovascularization, and pigmented anterior capsule. Slit lamp Exam of the left eye was normal. Dilated fundus exam of the right eye showed flat retina 360 degrees, bare RPE with surrounding laser scars. Dilated fundus exam of the left eye was normal.\nMaxitrol and Ofloxacin were discontinued, atropine dose was reduced to once daily, and patient was given a slow taper down of prednisolone eye drops over 3 weeks. Patient was advised again about the importance of face-down positioning.\nLater, the patient was released from jail, and he stopped following with our clinic. |
This case presentation has been consented by the family and approved by the ethics committee of South Medical University Affiliated Maternal & Child Health Hospital of Foshan.\nA 22-year-old G2P0 woman presented at weeks’ gestation for evaluation of a fetus with a left lung lesion. A detailed ultrasound examination considered pulmonary sequestration, but pulmonary cystadenoma could not be totally excluded. Serial ultrasonographic monitoring demonstrated an increasing size of the lesion of the left lung and the heart was shifted to the right. The ultrasound examination revealed a 5.6 cm × 5.0 cm × 5.0 cm mass at weeks’ gestation (Fig. ). The presentation was believed to be consistent with a CCAM. The cystic adenomatoid malformation volume ratio (CVR) was 2.8. Amniocentesis confirmed normal karyotype and no other anatomic abnormalities were present on detailed ultrasonographic survey.\nNonimmune hydrops were not found before operation, and the heart function was well preoperatively (Fig. ). Given the continued growth of the lung mass, the concern for the progression of hydrops and fetal demise, and the significant pulmonary hypoplasia believed incompatible with survival secondary to mass effect, multidisciplinary counseling was performed regarding open fetal surgery. After full evaluation and counseling by the fetal surgery team, the patient and her family wished to proceed with intrauterine surgery. Informed consent was obtained in accordance with the surgery protocol, and the open fetal surgery was approved by the ethics committee of South Medical University Affiliated Maternal & Child Health Hospital of Foshan.\nA maternal hysterotomy, left fetal thoracotomy and CCAM resection were performed at weeks’ gestation (May 15, 2011) under deep maternal general anesthesia.[ The open fetal surgery techniques were performed as described below.\nIndomethacin and antibiotics were given preoperatively, and ritodrine hydrochloride provided the necessary uterine relaxation. Sterile intraoperative sonography delineated both the fetal and placental position. The uterine incision (about 10 cm) was made at the edge of the placenta.[ The left fetal hand was delivered, intravenous access obtained, and a fetal pulse oximeter was placed. A left fetal thoracoabdominal incision was made based in the fifth intercostal space. Continuous fetal echocardiography assessed ongoing cardiac function and directed resuscitation requirements. After the thoracotomy, the mass was slowly delivered from the chest cavity and the CCAM resection was initiated. The fetal chest wall was closed and the fetus returned to the uterine cavity. An intrauterine dose of dexamethasone (10 mg) and ampicillin (4 g) were administered after the amniotic cavity was filled with warmed (37 °C) lactated Ringer solution. And the uterine and abdominal incisions were closed in layers. The mother tolerated the procedure well and was given atosiban and ritodrine hydrochloride after the surgery. Continuous fetal echocardiography confirmed recovery of fetal heart function after the intraoperative fetal resuscitation.\nPostoperatively, the mother was treated with intravenous magnesium sulfate, indocin, and transitioned to nifedipine for the remainder of the pregnancy to maintain uterine quiescence. The pathology from the fetal excision demonstrated pulmonary mass hyperplasia consistent with CCAM-like changes (Fig. ). The mother presented at weeks after open fetal surgery with preterm premature rupture of membranes (PPROM) and underwent cesarean delivery at weeks’ gestation. A vigorous woman infant of 1955 g (50th centile), with good Apgar score, was delivered. The baby's thoracoabdominal incision was well healed at birth without any evidence of fluid leakage. At 1 month, 4 years, and present, 5 years after birth, she has continued to do well without any obvious deficit and both respiration and circulation were well maintained (Fig. A, B).\nProgression of the patient's condition and accompanying interventions are illustrated in a flowchart (Fig. ). The surgery and follow-up are displayed in supplemental video. |
Patient 3: A 42-year-old female was admitted with back pain, gait disturbance, and moderate dysesthesia of right upper extremity. Symptoms were more apparent in the upper extremities than in the lower extremities. Assessment of neurological condition before surgery suggested Grade 3 on the modified McCormick functional schema and Grade 3 on the sensory pain scale. T2-weighted magnetic resonance images (MRIs) of the cervical spine showed local enlargement and intramedullary mixed signal of the spinal cord at C6 []. T2*-weighted gradient-echo MRIs showed mixed low signal within the spinal cord [], which was consistent with blood degradation products. Imaging diagnosis before surgery was intramedullary hemorrhage associated with cavernous malformation.\nThe patient underwent C5 to C7 osteoplastic laminotomy for evacuation of the intramedullary hemorrhage and total resection of the tumor. The spinal cord appeared slightly swollen []. Posterior spinal arteries on both sides were well differentiated from posterior spinal veins and the pial venous plexus []. Venous circulation of posterior spinal veins and the pial venous plexus appeared stagnant []. Crossing veins on the PMS were coagulated with a microbipolar coagulator at very low power levels under continuous saline irrigation. A PMS approach to the spinal cord revealed the intramedullary hemorrhage. Careful dissection within the spinal cord revealed the tumor–cord interface with the surrounding hemosiderin-stained tissue. The tumor was removed segmentally. The surrounding hemosiderin-stained tissue was not resected to avoid the damage of spinal tissue []. Posterior sulcal central veins were well visualized []. The pial edges were sutured together as much as possible to restore the shape of the spinal cord []. Supplemental digital content of surgical was provided.\nT2-weighted MRIs obtained 3 months after surgery showed satisfactory recovery of the intramedullary signal of the spinal cord []. Assessment of neurological condition at 3 months after surgery suggested Grade 2 on the modified McCormick functional schema and Grade 2 on the sensory pain scale. |
The patient’s course is outlined in Fig. .
A 58-year-old, previously healthy, white female presented to her primary care provider (PCP) with the desire to remove a right posterior scalp cyst for cosmesis. This non-inflamed, non-draining, painless, 1–2 cm cyst had been present for close to 10 years without change in size or fluctuance. The cyst was initially drained by the PCP, but, when it recurred 6 months later, the PCP excised the cyst and sent the specimen for routine pathology. The initial read of the tissue sample was high-grade invasive carcinoma with squamous features and arising in association with a PTT. Based on the pathology, the PCP referred the patient to a plastic surgeon for a more definitive excision of the lesion and repair of the defect. The lesion was excised with negative margins and pathology read as invasive high-grade squamous cell carcinoma (SCC). Eight months post resection, the lesion recurred locally along with a palpable right posterior cervical lymph node (LN). A positron emission tomography (PET) scan at that time demonstrated hypermetabolic activity in the posterior occiput and in a posterior neck LN. Fine needle aspiration (FNA) of both the primary scalp lesion and LN were completed, and pathology was reported as SCC, similar to the primary lesion.
With a working diagnosis of locally advanced SCC, the patient’s care was referred to a tertiary care center. There, a dermatopathologist re-evaluated the previous biopsy specimens and altered the diagnosis from SCC to MPTT (Fig. ). Subsequently, the patient was referred to a head and neck surgeon for modified radical posterior neck and lymph node dissection. Intraoperative findings uncovered the presence of nodal metastases to the posterior neck with extranodal extension, extensive perineural invasion of the spinal accessory nerve, and jugular venous invasion of the MPTT. After surgery, the case was discussed at a multi-disciplinary tumor board, and a common |
A 30-year-old housewife of Asian origin diagnosed to have pulmonary tuberculosis, was referred to spinal team as cauda equina lesion by her general practitioner. She was on antitubercular treatment (ATT) for last three months and, had history of progressive weakness in lower limbs for a week with urinary incontinence for a day. On initial examination she had stable airway, breathing and circulation. Neurological examination revealed increased tone and decreased power in both lower limbs (MRC 3/5). Sensory level was present at the level of D9. Knee and ankle reflexes were increased bilaterally with up-going planters. Anal tone and perianal sensations were decreased. On catheterization she had a residual volume of 400 ml. The clinical examination revealed a mixed picture. Whereas a sensory level at D9 with upper motor neuron signs in lower limbs (increased jerks and up-going planters) indicated a compressive lesion at D9, urinary retention along with decreased perianal sensations and, impaired anal tone and reflexes favoured a clinical diagnosis of cauda equina compression. Due to equivocal clinical signs initial impression was a multilevel disc disease with acute compression of cauda equina region. Urgent MR scan was done within 2 hours of clinical examination in order to rule out cauda equine lesion. On her way back from radiology department she dropped blood pressure and collapsed. Fluid resuscitation was started and re-examination confirmed large pulsatile mass in epigastrium with feeble femoral and dorsalis pedis pulses in both lower limbs. Lower extremities were cold on palpation; capillary refill was delayed to 3 sec with appreciable radio-femoral delay. Hypotension was resistant to fluid challenge and surgical team was contacted for advice. With the advice of vascular team a CT scan was requested. In view of her deteriorating clinical condition and low haemoglobin (8.5), decision to perform urgent laparotomy without CT scan was made for a possible leaking aortic aneurysm.\nMRI scan was reported in the mean time which showed the presence of massive pre-vertebral abscess in the thoracic region, pushing the aorta anteriorly resulting in significant narrowing of descending thoracic aorta (Fig ). The abscess was extending from D6 to D12 level measuring 12 × 18 × 20 cm in size (Fig &). The vertebral bodies of the D-7 to D-12 were severely destroyed, resulting in anterior angulation of thoracic spine. The spinal cord was compressed at the levels of D7/8 and D10/11 with signal changes (Fig ).\nDue to presence of the acute compression of the thoracic aorta and spinal cord an emergency surgery was performed by spinal team to relieve the compression, with vascular surgeons on standby. Spine was approached through anterior trans-thoracic route via sixth intercostal space. A big abscess compressing the aorta was encountered anterior to thoracic vertebrae extending from D6 to D-12. Aorta was severely compressed by the abscess with no evidence of pseudoaneurysm. The abscess was drained and sample was sent for microbiology. The neighbouring soft tissue was also sent for histopathological examination with excision and debridement of necrotic dead tissues. Femoral and dorsalis pedis pulses returned to normal on operating table soon after drainage of abscess. Keeping in view, the presence of a heavy infection, drains were left in situ, and the wound was closed without any in spinal stabilisation.\nAFB staining was positive for Mycobacterium tuberculosis from pus as well as surrounding soft tissue samples, which was confirmed on culture too. Poor patient compliance to previous ATT was discovered, which was restarted again post-operatively with the advice of microbiologist. She was started on Rifampicin, Isoniazid, Pyrazinamide and Ethambutol for initial 2 months followed by Isoniazid and Rifampicin for another 10 months. The power in the both lower limbs came back to normal on second postoperative day with resolution of urinary incontinence. She responded well to the treatment with improvement in her systemic symptoms in one week. The drain was taken out on second postoperative day. The patient was re-operated after six weeks, and spine was stabilised by plating and bone-grafting. The patient was gradually mobilised and discharged home after satisfactory recovery. The patient remained asymptomatic in three year's follow-up. Pulmonary tuberculosis which flared up initially also settled after recommencing the ATT. Follow-up x-ray revealed a satisfactory spinal fusion and well aligned prosthesis in situ. |
The patient is a 47-year-old Caucasian female who presented to the Emergency Department of an academic tertiary-care hospital in the Midwestern United States with complaint of left-sided weakness of the upper and lower extremities and right gaze preference three weeks after a right pontomedullary infarct complicated by Posterior Reversible Encephalopathy Syndrome (PRES) [that initial infarct had been treated in a different state]. Imaging revealed an acute infarct in the posterior limb of the right internal capsule without hemorrhagic transformation and an acute punctate infarct in the right parietal subcortical white matter with corresponding diffusion restrictions, as well as remote evidence of subcortical chronic diffuse microhemorrhages (). The Psychiatry Consultation & Liaison service was consulted on hospital day 2 after the patient reported, “I want to strangle myself with my oxygen cord.”\nOn initial evaluation, the patient reported history of anxiety treated previously by her primary care physician (PCP). She reported she had been frustrated with her medical condition but really did not intend to harm herself. She reported fluctuating mood since her initial stroke and had “good days and bad days.” She denied prior history of inpatient or outpatient psychiatric care or prior suicide attempts. She was oriented to person and place, but not time, was able to state the days of the week forwards, but not backwards, and endorsed visual hallucinations during her hospitalization. This presentation was felt to be consistent with delirium, and she was started on quetiapine 25 mg.\nFollowing a six-day medical admission, the patient was discharged to the acute inpatient rehabilitation unit housed within the hospital. Extensive diagnostic studies did not reveal an underlying etiology for the strokes, which were thought to be due to uncontrolled hypertension.\nPsychiatry was reconsulted by the rehab physicians for management of problematic behaviors. The patient exhibited ego-dystonic behaviors for which she would later apologize including repeatedly climbing out of bed, shouting for nursing assistance without clear need for help, shoving her fist into her mouth to induce vomiting, and periodic, purposeless screaming. These behaviors were disruptive to staff and other patients on the unit. While initially conceptualized as residual hyperactive delirium, her behaviors persisted and continued testing for underlying causes of delirium including electrolyte derangement, occult infection, new or evolving cerebrovascular event, or excess medication burden which were unrevealing\nAfter 60 days of acute rehab, she had reached maximal benefit of that intervention and continued exhibiting behaviors incompatible with nursing home disposition. The patient was then transferred to the university's geriatric psychiatry inpatient unit on an involuntary mental health commitment for behavioral management.\nIneffective medication trials prior to transfer included quetiapine (25 mg at bedtime and 25 mg several times daily as needed), mirtazapine (7.5 mg at bedtime), olanzapine (initial trial of 2.5 mg at bedtime and 2.5 mg several times daily as needed and a second trial of 15 mg and 2.5 mg several times daily as needed), buspirone (15 mg TID), divalproex (initial trial of 750 mg at bedtime and a second trial of 500 mg TID with lactulose and levocarnitine for hyperammonemia), melatonin (9 mg at bedtime), propranolol (40 mg QID), trazodone (150 mg at bedtime), gabapentin (200 mg several times daily as needed), dextromethorphan (20 mg BID, given as Robitussin), and clonazepam (0.5 mg AM and 1 mg PM).\nThroughout this period, the patient remained intermittently apologetic for her behaviors. Orientation was typically attuned to person, sometimes place, and generally not to month or year. She consistently denied depressed mood, anxiety, visual hallucinations, auditory hallucinations, paranoia, suicidal ideation, or homicidal ideation. Thought process remained concrete and perseverative with limited spontaneous speech output and paucity of thought content. Language remained intact without evidence of aphasia. Recent and remote memory were difficult to assess formally due to behavioral disturbance, but she had difficulty remembering recent details of her hospital course and remote details of her life prior to moving to her current city. She required staff assistance for completion of toileting, dressing, and feeding. She had deficiencies in executing complex motor tasks, such as getting out of bed, and was frequently found diagonal in bed with a limb tossed over the side-rail. These deficiencies were in excess of the residual motor effects of her strokes and suggestive of alterations in visuospatial skills, executive function, and planning. Her aberrant vocalizations did not appear goal-oriented and were not ameliorated by staff presence. This presentation persisted and was thought to represent a new cognitive baseline meeting diagnostic criteria for major vascular neurocognitive disorder with behavioral disturbance.\nNonpharmacological strategies including music, sensory stimulation, one to one time with staff, and frequent repositioning were tried without improvement in her symptoms. Additional ineffective medication trials following transfer to inpatient psych included fluoxetine (60 mg per day), retrial of dextromethorphan with fluoxetine as an enzymatic inhibitor (again to 20 mg BID), retrial of quetiapine (up to 600 mg total per day), haloperidol (5 mg several times daily as needed IM), oxycodone (5 QID), lorazepam (up to 6 mg daily), carbamazepine (200 TID), and chlorpromazine (50 QID). Throughout these trials, the patient continued to exhibit frequent periods of severe psychomotor agitation requiring vest restraint and purposeless screaming alternating with periods of oversedation following medications. Other than providing intermittent sedation, no particular combination of medications proved effective in treating the target symptoms.\nAt this point, having exhausted all reasonable behavioral and pharmacologic options, the inpatient psychiatric team recommended ECT as a last intervention prior to pursuing a palliative approach. Medical Ethics was consulted and felt ECT to be consistent with her previously articulated beliefs and wishes.\nThe patient was formally evaluated by the ECT service and, given her incapacity to consent, a court order was obtained for the procedure. She underwent an acute course of bitemporal ECT using a MECTA Spectrum 5000Q machine. She received methohexital and succinylcholine as anesthetic and relaxant agent, respectively. A dose-titration method was used to determine stimulus intensity. She received treatments at 50% over seizure threshold with the following parameters: pulse width: 1 millisecond, frequency: 20 Hz, duration: 2 sec. Treatments were given three times per week. She was maintained on chlorpromazine (50 mg QID) and lorazepam (1 mg QID) during the treatments. Following the sixth ECT treatment, the patient rarely engaged in purposeless yelling, and remained quiet most of the day, experienced normalization of her sleep wake cycle, but still exhibited purposeless movements and psychomotor agitation requiring a vest restraint at night.\nFollowing the third week of ECT treatments, she was consistently having low scores on the Pittsburgh Agitation Scale (PAS) and had minimal requirements for as needed medications for agitation []. While she still required a vest restraint overnight, her psychomotor agitation had improved dramatically. She resumed feeding herself with her right arm and tolerated pureed foods for the first time in six months. Following an acute course of 16 treatments, ECT was tapered to twice weekly and she started sertraline 25 mg in preparation for further decrease in ECT frequency. She remained stable and was successfully discharged to a nursing home with continuation of ECT as an outpatient. Following the expiration of the original court order for ECT, outpatient ECT was discontinued and the patient's family chose to not pursue a renewal of the order for continued treatment. She received 29 treatments in total. Nursing home staff reported that her behaviors remained in control after stopping ECT and she was thereafter able to return home with her parents. |
The second patient is an 82-year-old woman with knee osteoarthritis (OA) and no history of systemic inflammatory joint disease. In the right knee joint, apart from typical pain and limited mobility, the patient also suffered from recurrent effusion secondary to OA. The effusion was refractory to repeated joint punctures with intra-articular glucocorticoid administration. The patient did not consent to knee arthroplasty or arthroscopic joint debridement and, since she suffered from type 2 diabetes, numerous glucocorticoid injections made the patient’s blood glucose level difficult to control. Taking into consideration all available therapeutic options, the patient was referred for RSV as a supportive treatment.\nUnder US guidance, the suprapatellar recess of the right knee joint was punctured from the lateral approach. Approximately 40 ml of clear, yellowish fluid was evacuated, and 185 MBq of colloidal yttrium-90 (90Y) citrate was injected followed by 2 ml of sterile saline to flush the residual radiopharmaceutical from the needle. A sterile dressing was applied, the knee was immobilized, and the patient was advised not to walk or stand for at least 48 hours in order to minimize the risk of radioactive leakage. Thirty minutes after RSV, post-therapeutic positron emission tomography/computed tomography (PET/CT) with 90Y was performed (Biograph 64, Siemens) [], showing diffuse distribution of the radiopharmaceutical in the suprapatellar recess and medial knee compartment, with no evident extra-articular radioactivity ().\nDuring the follow-up visit after 4 weeks, the patient complained of a draining sinus with discharge of yellowish, turbid fluid and tender erythema at the site of the joint puncture (). US examination revealed that the sinus canal was 8 mm deep and was surrounded by hyperechoic tissue, with an increased power Doppler signal. Deeper layers of subcutaneous tissue were within normal limits, the articular capsule was intact and the wound did not communicate with the joint cavity. The patient admitted that she had not complied with the recommended knee immobilization and bed rest after RSV. Accordingly, the sinus was most likely the result of a partial radiopharmaceutical reflux along the needle tract into the subcutaneous tissue.\nMicrobiological cultures taken from the wound were negative. The consulting surgeon advised against surgical excision of the lesion, since poorly controlled diabetes and obesity could compromise the wound healing. A conservative approach was employed, including flushing the sinus with saline, debridement of necrotic tissue, application of hydrocortisone/oxytetracycline ointment and regular changes of the sterile dressing. Six months after the RSV, the fluid discharge and tenderness subsided, leaving a depression in the skin with scar tissue (). |
Eleven years ago, when our patient was a 33-year-old unmarried nulligravida, she developed CCC of the ovary for the first time. At that time, her menstrual cycle was regular and 30 days long, with dysmenorrhea, manifesting as lower abdominal and lumbar pain, persisting for 5–7 days. There was no family or medical history of note. She had visited a local clinic with the chief complaint of lumbar pain, where a right ovarian tumor 8 cm in size that included solid internal components, was identified, and she was referred to the University of Tsukuba Hospital. Transvaginal ultrasound had revealed a right adnexal mass with solid components, 68 mm × 53 mm in size, and an intramural uterine fibroid 23 mm × 27 mm, with no hypertrophy of the endometrium, which measured 2.1 mm. Blood counts and blood biochemistry test results revealed no abnormalities. Her serum CA19-9 level was 45.9 U/ml, CA125 was 22 U/ml, and CEA level was 1.3 ng/ml. Abdominal computed tomography (CT) and magnetic resonance imaging (MRI) revealed an 80-mm unilocular cystic mass in the right ovary with a papillary protrusion of longest diameter 45 mm. The left ovary was not enlarged, and there was no obvious peritoneal dissemination or enlarged lymph nodes. Surgery, comprising right adnexectomy, left ovarian biopsy, partial omentectomy and uterine fibroid removal, was performed. The right ovary was enlarged to 8 cm in size, and it was removed without intraoperative rupture and with its capsule intact. The left ovary was not enlarged, but was seen to have a small endometrial cyst, which was resected. The absence of peritoneal dissemination and enlarged lymph nodes was confirmed during surgery. The pathological diagnosis was CCC localized to the right ovary and endometriosis was observed in the left ovary, with ascites cytology Class III. The cytology was composed of clusters that include atypical cells having somewhat nuclear enlargement, and it makes a diagnosis difficult to distinguish mesothelial cells from malignant cells. The patient expressed a strong desire to preserve her fertility. Hence, pelvic and para-aortic lymph node dissections were performed via staged laparotomy, which revealed no metastases in any of the 91 pelvic or para-aortic lymph nodes that were removed, leading to a diagnosis of Stage IA CCC. Four courses of postoperative combination paclitaxel (175 mg/m2) and carboplatin (AUC6) chemotherapy (TC therapy) were administered to complete the initial treatment.\nNine years after the initial therapy, MRI during regular 6-monthly monitoring revealed the appearance of a 95-mm polycystic mass with a mural nodule of longest diameter 53 mm in the left adnexal region. Those findings were never seen at the previous MRI. At this time, her serum CA19-9 level was 12.9 U/ml, CA125 was 18.3 U/ml, and CEA level was 0.5 ng/ml, all of which were within normal limits. Disease recurrence in the contralateral ovary was diagnosed, and total abdominal hysterectomy, left adnexectomy and omental biopsy were performed. At the time of surgery, the left ovary was enlarged to 95 mm in size, and it was removed intact without intraoperative rupture of its capsule. The internal lumen of the tumor contained several milky-white mural nodules extending around 15 mm into the cavity (Fig. ). The histological diagnosis was CCC, but in addition to the possibility of recurrence, it was also considered that the tumor might have developed de novo, and the fact that clear cell adenofibroma (CCAF) was also present (Fig. ) suggested that this might have provided the genesis for its development. In addition, there were no endometriotic lesions in the non-solid cyst wall, and it only consists of fibrous membrane. The tumor was localized to the left ovary, and since ascites cytology was negative, it was diagnosed as Stage IA disease. The treatment was completed with four courses of postoperative TC therapy.\nTwo years after the completion of treatment, the patient is continuing outpatient monitoring with no sign of recurrence to date. |
A 36-year-old male with benign hypertension but otherwise no significant previous medical or surgical history presented to the emergency department with left gaze preference and right-side hemiparesis after being found down in his home with unclear time of onset of symptoms. CT angiography (CTA) revealed a distal left M1 middle cerebral artery (MCA) occlusion with an Alberta stroke program (ASPECTS) score of 9 on non-contrast CT of the head. Based on a favorable ASPECTS score, plans were made to proceed with endovascular mechanical embolectomy and the patient was transported to the neurointervention suite. Access to the right common femoral artery was obtained, and subsequent efforts were made to catheterize the left common carotid artery (LCCA). Initial attempts were unsuccessful at catheterizing the LCCA, and subsequent arch aortogram revealed that the patient had an aortic arch anatomy consistent with a congenital vascular rare variant of the great vessels. Volume-rendered 3D images from a concurrent CTA chest examination revealed a right aortic arch with the LCCA arising from the ascending aorta, as well as aberrant course of the brachiocephalic artery (Figure ). Several consecutive attempts to catheterize the LCCA were unsuccessful due to unfavorable trajectories off the aortic arch, at which point it was decided to proceed with an alternative access route. Given the aforementioned anatomical variant, it was unlikely that a transradial approach to the LCCA would be successful; thus, it was decided to perform a direct left common carotid artery puncture to gain intracranial access.\nThe left neck was prepared and draped, and a suitable puncture site was confirmed. After administration of 1% lidocaine, ultrasound-guided access to the left common carotid artery was gained (Figure ). A 6-French 10-cm sheath was placed at this time. After ensuring proper placement in the left MCA, endovascular mechanical embolectomy was performed with a 4 mm x 20 mm Solitaire stentreiver device (Medtronic Inc, Minneapolis, MN, USA) in conjunction with aspiration with Penumbra ACE68 catheter (Penumbra Inc, Alemeda, CA, USA) (Figure ). At the conclusion of the procedure, the LCCA arteriotomy site with closed by the vascular surgery team via open surgical repair. The patient subsequently recovered remarkably well at post-procedure follow-up with a three-month modified Rankin score of 1. |
A 22-year-old male with a past medical history of pericarditis and pericardial effusion presented to the ED with the chief complaint of facial swelling, which had been present for the prior three weeks. The swelling was predominantly on the right side of his face and upper lip. He had no history of angioedema, had not started any new medications, and was not aware of an environmental exposure that immediately preceded the onset of swelling. In addition to the facial and lip swelling, the patient reported a rash of the same duration on his chest and shoulders. Additional associated symptoms included decreased exercise tolerance, exertional dyspnea, and a single episode of dark, maroon-colored stool. He denied fever, chills, myalgia, arthralgia, chest pain, abdominal pain, nausea, vomiting, odynophagia, dysphagia, and confusion. He was not aware of any sick contacts and he had not traveled recently. He reported that his family did not have a history of chronic illnesses.\nPhysical examination was significant for a blood pressure of 104/58 millimeters of mercury, a pulse of 96 beats per minute, respiratory rate of 16 breaths per minute, a temperature of 36.8° Celsius, and a pulse oximetry reading of 100% on room air. He was a thin young man who did not appear to be in distress or acutely ill. Bilateral facial edema along with edema of the upper lip was noted (). In addition, his conjunctiva, palms, and soles were notable for pallor. A petechial rash was observed on his upper chest, bilateral shoulders, tongue, and soft palate (). A malar rash was also noted (). The remainder of his examination was normal.\nHis initial ED evaluation included a chest radiograph, electrocardiogram, and laboratory studies. The results of pertinent laboratory studies are listed in the . Given his severe thrombocytopenia and anemia, thrombotic thrombocytopenic purpura (TTP) was considered and an emergent hematology consultation was obtained. A peripheral blood smear demonstrated 1–2 schistocytes per high-power field, which initially raised concern for a microangiopathic hemolytic anemia. As a result, a hemodialysis catheter was inserted and plasmapheresis was initiated while the patient was in the ED. He received a unit of packed red blood cells along with corticosteroids and was admitted to the medical intermediate care unit.\nWorkup revealed a positive immunoglobulin G (IgG) Coombs test. He also had a high titer of antinuclear acid antibody and low C3/C4 complements, indicative of an acute exacerbation of an autoimmune disease. The combination of his symptoms, ED workup, and history of pericarditis and pericardial effusion favored the diagnosis of systemic lupus erythematosus (SLE). Within 48 hours after admission, an A disintegrin and metalloproteinase with thrombospondin motifs 13 (ADAMTS13) level returned with 78% activity and less than 5% inhibitor. This result was not consistent with the diagnosis of TTP, and plasma exchange was stopped. Ultimately, the hematologist diagnosed Evans syndrome as a presenting feature of SLE. |
A 56-year-old female presented with a four-week history of increasing shortness of breath, cough, and mild dysphonia. She had been treated with antibiotics for chest X-ray demonstrated pneumonia during this time but received little benefit. On examination she was found to be “wheezy” on auscultation of the chest and she was admitted under the respiratory service.\nA new diagnosis of asthma was suggested for which she was started on salbutamol nebulizers and oral prednisone forty milligrams. She made little progress and suffered sporadic episodes of oxygen desaturation. On day nine of admission, a computed tomography scan of the chest demonstrated known pulmonary metastases and an irregular appearance of the larynx. On the tenth day of admission, an otolaryngology consult was sought.\nThe patient's medical history was significant for locally advanced, Stage IIIb (T4, N1, M0), adenocarcinoma of the rectum which was diagnosed and treated eight years prior to this episode. Adenocarcinoma treatment involved chemoradiation with Capecitabine as an adjunct for abdominoperineal excision of the rectum with vertical rectus abdominis myocutaneous flap. Four years after her initial treatment she required radio-frequency ablation for lung and liver metastases, she underwent a partial right lung resection five years after initial treatment, and six years after initial treatment she required further radio-frequency ablation of liver and lung metastases. Her metastases proved resistant to radio-frequency ablation and at the time of admission she had received four cycles of palliative Capecitabine and Mitomycin chemotherapy. She had suffered two pulmonary emboli, one six years before this episode and one six months prior, for which she took regular prophylactic low molecular weight heparin.\nOn otolaryngology review, she had biphasic stridor on deep breathing. Flexible nasendoscopy (FNE) was performed at the bedside which visualized a large nodular subglottic mass. Epinephrine nebulizers and intravenous dexamethasone were initiated. On the day of review, a head and neck operative list was taking place. Between cases, a specialist head and neck otolaryngologist and a specialist “difficult airway” anesthesiologist reviewed the patient with repeat FNE. A decision was quickly made to add the patient to their operative list for surgical resection of the mass while securing the airway.\nIntubation was performed with the assistance of the Karl Storz C-MAC video laryngoscope and an endotracheal tube was “railroaded” beyond the subglottic mass with an Eschmann tracheal tube introducer. Microlaryngoscopy demonstrated a large sessile subglottic mass arising from the posterior commissure (). The bulk of this mass was easily removed en masse by a laryngeal grasper. After a surgical pause to assess for bleeding, the endotracheal tube was replaced with jet ventilation to allow for laser resection of the mass remnant and its underlying mucosa; ten watts of carbon dioxide laser was used on super-pulse mode for the resection. Following laser resection the tissue was extremely friable and bleeding followed any contact (). Therefore, in an effort to ensure no further bleeding into the airway, Tisseel fibrin glue (Baxter AG, Vienna, Austria) was applied over the site of laser ablation through a catheter inserted in the operative channel of the bronchoscope with the endoscopic applicator provided by the manufacturer. During Tisseel application, jet ventilation was held for between ninety seconds and two minutes. Two application cycles of Tisseel were employed with five minutes between applications (). Five minutes after the second application of Tisseel cessation of anesthesia was initiated (supplementary video online in Supplementary Material available online at ).\nThe mass measured twelve millimeters in its longest diameter and was histologically described as moderately differentiated adenocarcinoma morphological consistent with metastatic colorectal carcinoma, with positive expression of Cytokeratin 20 and Caudal Type Homeobox 2 without Cluster of Differentiation 7. Immunohistochemical studies for mismatch repair proteins found no evidence to support a diagnosis of Lynch syndrome. Pathological analysis is therefore strongly suggestive of primary tumor origin in the large intestine, confirming this mass as a metastasis of the patient's rectal adenocarcinoma and not a primary laryngeal tumor.\nThe patient was closely monitored for forty-eight hours on the high dependency unit with clear instructions to intubate if there was evidence of bleed into the airway. Subsequently she was stepped-down to the ward where significant resolution of her shortness of breath and cough were reported, and oxygen saturations above 96% were maintained throughout. Chest auscultation did not elicit any wheeze. Postoperative speech and language therapy evaluation found excellent speech and swallow function, and the patient was discharged from the speech and language therapy service prior to discharge. She was discharged from hospital one week after the procedure having had an uncomplicated postoperative course. Nine months postoperatively she was clinically stable, without evidence of tumor recurrence within the larynx. |
A 28-year-old male presented to the emergency department after exposure to an industrial blast injury, involving the chest and anterior abdominal wall. He was unconscious with labored breathing and pain. His Glasgow Coma Scale deteriorated to 8, and a definitive airway was placed without any incident. Positive pressure ventilation was instituted. The primary survey showed decreased air entry on the left side of the chest. All peripheral pulses were palpable and symmetrical on initial presentation.\nChest radiograph showed the left side chest opacification indicating hemothorax []. Thoracostomy was performed with an initial gush 450 ml of dark blood. Pericardial focused assessment with sonography for trauma was equivocal, and chest X-ray demonstrated a failure to fully evacuate the hemothorax.\nThe secondary survey showed superficial left hand, thigh, and leg cut wounds. Pieces of shrapnel were embedded in the left arm, anterior chest, and abdominal wall. He was stabilized and had CT of the brain, cervical spine, chest and abdomen. CT of the brain and cervical spine were normal. CT of the chest revealed fractures of the left first and second ribs. A hyperdense foreign body was seen penetrating the anterior chest wall soft tissue guarded by ribs without deep penetration to the heart []. There was evidence of retrosternal soft tissue hematoma and left-sided hemopneumothorax with underlying pulmonary contusion. Mild hemopericardium with air was seen within the pericardial sac but no significant major vessel injury. The left lateral chest wall and anterior wall surgical emphysema was also seen. CT of the abdomen [] showed pellets within the anterior abdominal wall with related surgical emphysema but no evidence of peritoneal penetration. A transthoracic echocardiogram demonstrated mild pneumohemopericardium without tamponade.\nRepeat examination of the lower limb revealed a palpable diminution in the right dorsalis pedis pulsation. Ankle-brachial pressure index was performed which revealed a significant discrepancy between the two lower limbs (right 0.4 vs. left 1.1). Review of the original CT revealed evidence of a foreign body in the right groin with no clear point of entry and a lack of subcutaneous air. With further deterioration in the status lower limb pulsation, he was shifted for CT angiography which revealed a metallic foreign body in proximity to the right common femoral artery (CFA) masking its bifurcation point with streaky artifact, just a few millimeters distally. A filling defect was noted within the superficial femoral with no pseudoaneurysm. Although there was no subsequent drop in his systolic blood pressure, an arterial line tracing revealed evidence of pulsus paradoxus.\nA joint decision was made to proceed with an emergency median sternotomy and right femoral artery exploration by the cardiac, trauma, and vascular surgery services. Intraoperatively, blood in pericardium with clots was evacuated. There was a large laceration about 4 cm in the inferior surface of right ventricle without active bleeding and just opposite, a 4 cm tear in the diaphragm tendon.\nCardiorrhaphy with pledgeted sutures and diaphragm repair was performed. The left pleura was opened, and clots were evacuated. Transesophageal echocardiography was performed intraoperatively, and no ventricular septal defect (VSD) was visualized. Shrapnel, lodged in the anterior chest wall muscles, was removed. Simultaneously, right groin exploration was performed. The proximal, distal, and profunda femoris were individually controlled. The foreign material was felt and removed from the CFA at the bifurcation by means of an arteriotomy []. A 4F Fogarty catheter was passed to 60 cm with good backflow and return of thrombus and debris. Right, lower limb pulses were regained. A primary repair of the artery was performed.\nThe patient was shifted to the Cardiac Intensive Care Unit in stable condition. His hemodynamics was normal without inotropic support. His lower limb pulses remained palpable. On the second postoperative day, upper limb numbness was noted, and a CT arteriogram of the head and neck was performed to rule out further embolic disease in the distribution of the carotid artery; this was negative. The patient did well in the following days and was discharged with no complications. He presented to the clinic for removal of symptomatic-retained foreign bodies from the hand and anterior abdominal wall which was successfully extracted and with further follow up he was recovered totally. |
Nine-year-old pediatric girl patient came to the emergency department with the history of an Allen key injury on her left cheek (). The family of the patient said that when the patient was traveling in the car, Allen key in her hand was inserted into her face because of the sudden brake.\nOn examination, an entry of foreign body was found on the upper left cheek. The tip of Allen key was seen at nasopharynx with diagnostic nasal endoscopy after the nasal decongestion. There were edema and tenderness over the left cheek. It revealed no hemorrhage of the turbinates or the nasal mucosa on anterior rhinoscopy. Firstly the lateral skull roentgenogram revealed Allen key in the left maxillary sinus to nasopharynx. The body of the Allen key has a very close relation to the floor of the orbit and the sphenoid sinus (). This image was good enough for us, but the computed tomography (CT) was also used to make sure that the periorbital and skull base were damaged or not. CT of the maxillofacial region revealed no orbital and intracranial injury (). The patient also had no neurological or vascular injury.\nThe patient was immediately operated on under general anesthesia. During the operation, the foreign body was removed after short-term cauterization with monopolar cautery due to its metallic nature by making a millimetric incision on the adjacent skin tissue to prevent skin damage (). This step was done to prevent active bleeding. The entry site and incision were sutured when no hemorrhage was seen after the foreign body was removed. The left nasal passages were again examined by nasal endoscopy and no abnormality was detected. Antibiotics were given for seven days to prevent possible infection. The patient was discharged on the same day of the operation.\nAt the 6-month follow-up, the patients' physical examination and radiological investigations were normal. |
A right total hip revision was performed in a 56-year-old woman for aseptic loosening of the primary THA. The primary arthroplasty was implanted 14 years before that for rheumatoid arthritis. During the revision surgery, a Burch-Schneider metal cage was used to treat the acetabular bone defect. During the first postoperative week, a typical right (homolateral) sciatalgic pain developed in association with a severe lumbar back pain. The position with the knee flexed of approximately 30° provided moderate pain relief. The Tinel, Lasegue, and Valleix tests were all positive on the right side. After six days of pharmacological treatment, the sciatalgic pain was evaluated with a magnetic resonance (MR) which demonstrated L1-L2 and L3-L4 herniated discs, but no specific radicular compression.The radiological evaluation of the prosthesis did not show mobilization or malpositioning of the implant (). After approximately ten days, a palsy of moderate grade of the extensor hallucis longus (EHL) and of the extensor digitorum communis (EDC) arose in association with a low-grade hypoesthesia of the posterior plantar region of the right foot. The electromyograph test performed after the pharmacological treatment showed a high-grade delay of the nerve impulse at gluteal level. The hypothesis was of a compression of the sciatic nerve directly on the cage by a fibrotic scar tissue. Therefore, revision surgery and debridement of the sciatic nerve at the hip were planned. The posterior approach, following the previous exposure, was used. After the section of the tendon of the external rotator muscles group and of the piriformis muscle, the nerve appeared clearly embedded in a fibrotic tissue (). Moreover, a straight relationship between the sciatic nerve and the Schneider ring was found (): this relationship caused the compression of the nerve itself on the metallic cage, particularly during the movement of flexion of the hip, which clearly stretched the nerve. No anatomical alterations of the sciatic nerve through the piriformis muscle were observed. The Schneider cage appeared well positioned, so it was not necessary to revise the ring. The nerve was carefully released from the fibrotic tissue, and a muscular flap [] was made using part of the dissected profundus gluteus muscle to allow a nerve glide on the ischiatic bone and on the edge of the Burch Schneider metal cage (). During the second day after the operation, the patient had pain relief, improvement of the palsy, and the possibility to move the hip without pain (negative Tinel and Lasegue tests). After 45 days, the patient was reevaluated: the hypoesthesia and the palsies were completely cleared up. |
An 18-years-old Caucasian female referred to the practice with complaint of excessive gum display while smiling ( and ).\nThe patient presented a good general health and absence of active periodontal disease. Clinically, the patient had short clinical crowns and a gummy smile showing gingival exposure greater than 3mm ().\nNo periapical radiolucency at radiographic examination was detected, the periodontal ligament was within normal limit, and the crown-to-root ratio was about 1 : 3. At clinical examination,\nattached gingiva band was 6 to 7mm in width, and periodontal pocket depth was 3mm or less.\nThe primary treatment plan proposed was a combined orthodontic-surgical approach by mean of LeFort I osteotomy in order to affect the maxilla and reduce the gingival exposure. The patient refused the\nsurgical approach. A second treatment plan was developed and proposed consisting of a preliminary gingival surgery (gingivectomy) followed by injection of botulinum toxin type A. The patient was informed\nabout the recurrence of gummy smile after 6 months of application because of temporary results of the botulinum toxin. The patient read and signed a written consent form prior to treatment. Under local\ninfiltrative anesthesia, bleeding points were determined with the aid of a millimetered- probe and the union of these points were performed with the electrocautery.[]\nThe length of the teeth was increased, characterizing the dental zenith. Subsequently, the scraping was performed, resembling the technique of external bevel, in order to enhance issue healing\n( and ). There was no need of the use of surgical cement, given that the process of the wound occurs by secondary\nintention. The patient reported no complaints or complications after surgery.\nAfter 30 days, at the subsequent consultation, satisfactory tissue repair was observed (), and no changes or complaints were reported by the patient.\nHowever, the persistence of complaint of gummy smile was reported by the patient ( and ).\nIn the same consultation, botulinum toxin was applied. Prior to application of botulinum toxin, the surface of the skin was disinfected with ethyl alcohol 70% and the oils from the area were removed, in order to avoid local infection. The points of application were marked, beside each nostril. Then, local anesthetic (EmlaTM, Astra, São Paulo, Brazil) was applied with the aim of promoting comfort during the procedure. Botulinum toxin type A (DysportTM, Ipsen Biopharm Ltd, Wrexham, UK) was diluted in 1.7ml of saline according to the manufacturer's instructions, and two units was injected in the recommended site, laterally to each nostril. After application, the patient was advised not to bow their head during the first four hours and not engage in physical activity during the first 24 hours after the procedure.\nAfter 10 days, the patient was examined. She presented a uniform dehiscence of the upper lip and reduction and attenuation of gummy smile ( and ). Side effects or complaints were not reported. |
A 16-year-old Indian boy was brought to our hospital with swelling in the midline lower back that had been present since birth (Figure ). The swelling had gradually increased to its size at presentation and was associated with mild physical discomfort. Apart from these findings, there was no significant history as far as the patient's swelling was concerned.\nThe initial examination revealed a cystic, non-mobile, non-tender mass approximately 15 cm × 15 cm in size attached to the back in the midline in the lumbosacral region. However, the patient's blood counts, urine analysis, and liver function test results were normal. Furthermore, the radiographs of the lumbosacral region showed a well-defined swelling 15 cm × 20 cm in size with a smooth margin from the L3 vertebra to the S3 vertebra (Figure ). On the basis of our clinical suspicion of a cystic tumor, fine-needle aspiration cytology (FNAC) was performed to confirm the diagnosis. The results were positive for a mature cystic teratoma. Accordingly, the patient was prepared for surgery, and MRI was performed to establish the extent of the tumor. MRI of the lumbosacral spine revealed a well-defined lesion in the midline extending to the right gluteal region in the subcutaneous plane from approximately the L3-L4 to the S4 vertebrae and crossing the midline. It was further observed that the tumor was hyperintense on T1-weighted images and hypointense on T2-weighted images, which was suggestive of fat contents. There was no obvious communication with the spinal cord (Figure ).\nThe tumor was excised by creating an elliptical incision over the cyst. A whitish yellow, well-encapsulated, non-mobile mass was observed. The tumor was carefully dissected to allow us to reach the base, which was found to be attached to the L5 lumbar vertebra. The attachments, along with a small piece of lumbar vertebra, were also removed to minimize the chance of recurrence. |
Patient K., a 39-year-old female was admitted to a general hospital in Severodonetsk (Ukraine) on 14 December 2015 with complaints of severe lower abdominal pain radiating to the lumbar region, as well as difficulty urinating.\nShe had a history of Cesarean sections in 2003 and 2005, respectively. The latest Cesarean section ended up with iatrogenic injury to the apex of the bladder. The defects of the bladder and uterus were repaired separately. The patient was in satisfactory condition when discharged from the department of obstetrics.\nHowever, after three months the patient developed severe abdominal pain at the end of urination associated with scanty periods accompanied by hematuria. On the basis of characteristic symptoms vesicouterine fistula was suspected. This diagnosis was confirmed by cystoscopy. The patient was offered surgery, which she refused. Subsequently, due to resolution of symptoms the patient stopped seeking medical attention, although she periodically suffered from urinary incontinence, menouria, and recurrent infection of lower urinary tract.\nAfter examination the patient was diagnosed with missed abortion of ectopic pregnancy at 11 weeks of gestation localized in the bladder. The fetus in the cavity of the urinary bladder was detected at ultrasound examination (). The vesicouterine fistula was also well visualized. For further examination the patient was referred to Kharkiv Regional Clinical Perinatal Center and afterwards was transferred for treatment to Regional Clinical Hospital, Center of Emergency Medical Care and Disaster Medicine, Kharkiv Regional Perinatal Center.\nOn 15 December 2015 the patient underwent cystoscopy, which revealed in the cavity of the urinary bladder a dead fetus, embryonic vesicle, and fragments of the umbilical cord (). On the posterosuperior wall of the bladder a deck-edged mouth of fistula 2.0 cm in diameter was visualized.\nOn 17 December 2015 a joint team of urologists and obstetrician-gynecologists performed the following surgical treatment: laparotomy, vesicouterine fistula excision, removal of the fetus from the bladder cavity, bladder defect closure, and supravaginal amputation of uterus with removal of both fallopian tubes.\nIn the course of the operation some severe adhesions were found between the bladder, uterine body, and the parietal peritoneum. The uterus measured 11.0 × 9.5 × 10.0 cm, with pale pink color, softish consistency, and smooth outer surface. The uterus had limited mobility due to adhesions. Using the method of sharp dissection the bladder was separated from the anterior surface of the uterus toward the level of the fistulous tract. The bladder was opened. The body of macerated fetus was removed from the bladder cavity along with fetal membranes with clear signs of autolysis (). The bladder was circumferentially dissected away from the uterus around the fistulous tract (O'Connor's technique) (). The bladder was closed with Vicryl locking sutures, and Foley catheter was inserted into the bladder cavity through the urethra. Finally, supracervical hysterectomy was performed.\nThe postoperative period was uneventful. Patient was discharged from the hospital at the 8th day. The urethral catheter was removed 14 days after surgery. |
Patient 3 was a 16 year old girl that first presented in our outpatient clinic at 12 years of age. She had been born preterm by emergency caesarean section due to pre-eclampsia. Respiratory support through CPAP (“continuous positive airway pressure”) was needed for 4 days as well as a nasogastric tube. After discharge, new-born development was normal until the fourth month of life, when axial hypotonia was first noticed. At 12 months, discrete dyskinetic movements began. Developmental milestones were only met partially and with delay: at the age of 2 years, she was able to crawl and at 2.5 years, she was able to walk unassisted yet in a clumsy way and with need for a wheel-chair for longer distances. With 2 years, first hyperkinetic exacerbations appeared, lasting up to 1 min and occurring several times per hour. These hyperkinetic attacks started suddenly, were triggered by strong emotions, movement, temperature changes (for instance during bathing) and occurred rarely at night. During childhood, these sudden movement spells increased in frequency. Other clinical features such as hypersalivation, dysarthria and oral/tongue dyskinesia were reported. There were no signs of cognitive impairment. Family history was negative for movement disorders. Extensive clinical work-ups prior to first visit at our outpatient clinic including cerebral MRI at 1 and 5 years of age, analysis of cerebral spinal fluid including neurotransmitter metabolism and screening for metabolic disorders had remained elusive. Genetic analyses for dystonia, paroxysmal dyskinesia or episodic ataxia were negative but revealed a compound heterozygote ATM mutation of unknown significance that was first considered responsible for the disorder. Re-evaluation at the age of 12 revealed the pathogenic ADCY5 mutation. Treatment with levodopa was initiated at age of two (13 mg/kg/day) and led to an increase of dyskinesia. Carnitine, carbamazepine and clonazepam had no effect on the movement disorder.\nClinical examination at first visit showed a slim, pre-pubertal 12-year-old girl with permanent generalized choreoathetosis that was superimposed by intermittent myoclonus and dystonia. Axial hypotonia with dropped head was evident mainly when trying to stand up and during ambulation. She could stand unsupported for a few seconds. She would walk > 10 m without aid in a clumsy and dystonic manner. Perioral and tongue dyskinesia were nearly permanent and comprised slow choreatic movements as well as short jerks. Speech was effortful and dysarthric. Even though there was no apparent facial palsy, voluntary control of facial mimics and tongue movement were impaired. Deep tendon reflexes were brisk, pyramidal signs were negative. Limb muscle tone was normal, muscle strength fulfilled 5/5 MRC (“Medical Research Council” Scale). During the exam, the movement disorder exacerbated for episodes of < 1 min duration without an obvious trigger. The patient was not taking any medication at that time.\nAt the age of 13, DBS electrodes were implanted bilaterally in the GPi and a rechargeable Boston Scientific Vercise RC impulse generator subcutaneously in the left upper thorax. Immediately after surgery a reduction of dyskinesia was observed and sustained for about 6–8 weeks. 3 months after surgery, monopolar review was performed and new parameters were set, leading to a decrease in the hyperkinetic baseline movement disorder as well as the episodic storms. More than that, the ability to sit freely and to walk as well as the involuntary tongue movements and voluntary tongue control were improved. There was no effect on hypersalivation or dysarthria. 2 years after implantation the patient reported improved gait as biggest achievement of the stimulation. Furthermore, sitting and standing as well as tongue and facial dyskinesia were reported with clinically meaningful improvement. The sudden attacks during the day had become rarer. At the last follow-up visit, 3 years after implantation, the baseline movement disorder had deteriorated slightly, which resolved after subtle changes in current settings.\nRetrospective video ratings of the AIMS revealed a mild improvement of involuntary movements, no evident improvement was assessed by the BFMDRS (see Table for details). The girl and her father reported a subjective general improvement of 40–50% after DBS. 1 year after surgery, the battery of the IPG emptied completely due to technical problems. With delay of about 1 week involuntary movements increased leading to a visit of the emergency department. The transient worsening of the movement disorder resolved after proper charging of the IPG. |
A 32-year-old male was referred from a local clinic to our gastrointestinal department with complaints of dizziness and abdominal bloating. Despite 2 months of conservative treatment for abdominal discomfort, his condition had worsened. Endoscopy and abdominal sonography performed in the local clinic showed no specific findings and a huge mass in the cecal area, respectively. The cecal mass was evaluated by using abdominal computed tomography (CT), colonoscopy, and laboratory studies on admission to our hospital. Initial laboratory data showed 5.2 g/dL of hemoglobin and elevated levels of carcinoembryonic antigen (6.96 ng/mL). Colonoscopy revealed a large fungating mass with a tendency to bleed easily, which interrupted the lumen of the ascending colon (). Contrast enhanced CT of the abdomen showed invagination of the terminal ileum into the ascending colon with an ileocecal mass of 3 cm in size (), suggestive of ileocecal intussusception due to ileocecal cancer with multiple metastatic mesenteric lymph nodes (). Because the histology report based on a biopsy sample was a moderately differentiated adenocarcinoma with no other metastatic lesion noted except for several mesenteric and aortocaval lymph nodes on positron emission tomography-CT (PET/CT), the patient underwent a right hemicolectomy on the 7th day of admission.\nGross inspection of the lesion revealed a polypoid and ulceroinfiltrative growth measuring 8.0 cm × 5.5 cm in the cecum (). The mesocolon was macroscopically intact. The tumor involvement was approximately 70% of the circumference. The tumor invasion depth was to the subserosa. The proximal and the distal resection margins of the specimen were tumor free. Eleven of 49 lymph nodes were metastatic. Sheets of monomorphic large cells with large rounded nuclei and abundant cytoplasm were found on histopathologic examination of the cecal mass. Furthermore, islands of tumor cells with glandular structures accounted for more than 30% of the tumor (). Immunohistochemical examination revealed synaptophysin (strongly positive) and reactivity in the neuroendocrine differentiation area (). The final histopathological diagnosis was a high-grade MANEC of the ascending colon, tumor stage T3N2M0.\nThe patient underwent adjuvant chemotherapy. Adjuvant chemotherapy with 5FU and oxaliplatin (FOLFOX) was planned for six months, and the patient has undergone eight rounds of chemotherapy. The follow-up abdominal CT after four rounds of chemotherapy showed no evidence of tumor recurrence. |
A 60-year-old male with metastatic RCC treated with nivolumab and palliative radiation therapy presented to our institution in 2016 with shortness of breath and was found to be in acute respiratory failure. Computed tomography (CT) of the chest was significant for multiple new ground-glass opacities throughout bilateral lungs concerning for therapy-induced pneumonitis (Figures , ). The etiology of ground glass opacities includes but is not limited to infectious pneumonitis, bronchioloalveolar carcinoma, or interstitial disease. Given the timing of symptom onset as well as lack of response to infectious treatment, therapy-induced pneumonitis remained high on our differential.\nHe initially presented in 2011 with gross hematuria and right-sided flank pain and underwent right radical nephrectomy and lymph node dissection of a 9 cm Fuhrman grade IV RCC with negative margins and lymph nodes. Two years later, surveillance imaging and biopsy were significant for metastatic RCC in the lungs. He was initially treated with one year of sunitinib, a multi-targeted receptor tyrosine kinase inhibitor. However, given the progression of disease, he was transitioned to one year of pazopanib followed by six months of axitinib, one month of everolimus, and five months of sorafenib. Pazopanib, axitinib, and sorafenib are also tyrosine kinase inhibitors. Everolimus is an inhibitor of mammalian target of rapamycin. Given the lack of response to these therapies, our patient was started on nivolumab at 3 mg/kg in May of 2016. Over the course of four years, he received targeted palliative radiotherapy including 1900 centigray (cGy) to a left upper lobe lung mass in May 2016 and 800 cGy to an L5 lesion in September 2016.\nHe complained of chronic shortness of breath for three months felt secondary to anemia and a left pleural effusion before presenting to our hospital in acute respiratory failure with CT evidence of new diffuse ground-glass opacities occupying the majority of both lungs (Figures , ). Given high suspicion for therapy-induced pneumonitis, he was started on a treatment course of high dose steroids. However, the patient’s respiratory status continued to decline and he passed away on comfort measures.\nPathology was significant for organizing diffuse alveolar damage with hyaline membrane formation in all lobes of both lungs away from the metastatic RCC (Figures , ). There was no evidence of an infectious process from cultures and pathologic evaluation. This histologic reaction pattern is a typical finding in patients with a clinical diagnosis of acute respiratory distress syndrome (ARDS) concerning for therapy-induced pneumonitis. |
A 33-year-old woman (gravida 0, para 0) was referred to Osaka Central Hospital (Osaka, Japan) for gynecological, fertility-preserving laparoscopic surgery. According to her surgical history, two prior laparoscopic surgeries had been performed to treat endometriosis. At the age of 24, she underwent her first laparoscopic cystectomy for left ovarian endometrioma at a community hospital. When she was 29 years old, a second laparoscopic surgery (laparoscopic cystectomy for a right ovarian cyst) was performed at the same institution. The histological examinations of both specimens revealed benign endometriotic cysts. However, approximately 7 months following the second surgery, ultrasonographic and pelvic examinations revealed bilateral ovarian cysts. In response, we administered gonadotropin-releasing hormone agonist therapy (buserelin acetate, 900 μg/day) every 6 months. Upon termination of treatment, the cysts gradually enlarged. At 33 years of age, our patient got married. She desired children and was therefore referred to our institution for fertility treatment via laparoscopic surgery.\nUpon presentation to our hospital, both her ovaries were enlarged (left, 3 cm; right, 4 cm) because of endometrioma. MRI and ultrasound scans showed no solid component within the bilateral cysts. Initially, we considered terminating the hormonal therapy to allow for natural pregnancy. However, she did not become pregnant during a follow-up period of approximately 5 months. Therefore, our patient consented to a third laparoscopic procedure to improve her fertility.\nA preoperative MRI scan revealed a solid 2-cm component of her right ovarian endometrioma. Although the imaging was not enhanced, the possibility of malignancy could not be overlooked. Therefore, we recommended exploratory laparotomy to be performed at another institution. However, the patient and her husband rejected this recommendation and strongly insisted upon a third laparoscopic surgery at our institution. Our patient was fully informed of the treatment options and submitted written consent after which we agreed to perform the third laparoscopic surgery.\nNotably, the third laparoscopic surgery was performed at our institution approximately 10 years after the first surgery. The patient's cancer antigen 125 serum level was within the normal range and both ovaries, which were strongly adhered to surrounding tissues, were enlarged (right, 5 cm; left, 4 cm). The cul-de-sac was obliterated with dense pelvic adhesions. Laparoscopic cystectomy for both ovarian cysts and adhesiolysis were performed.\nThe patient had an uneventful recovery. A histological examination revealed a transition between the endometrioid adenocarcinoma and the directly adjacent endometriosis (fig. ). The cytological result of the small amount of ascites was negative. We reviewed the external slides of the previous two surgeries and confirmed the diagnosis of atypical endometriosis based on the second specimen (fig. ). The patient was diagnosed as having FIGO stage 1c(2), grade 1 ovarian endometrioid adenocarcinoma. Once informed of our diagnosis, our patient desired to attempt to preserve her fertility and rejected complete curative surgery for ovarian cancer.\nFor the fourth surgery, we performed laparoscopic salpingo-oophorectomy of the right ovary, biopsy of the left ovary and partial omentectomy. All specimens showed no pathological malignancy. Postoperatively, the patient underwent three courses of chemotherapy consisting of 175 mg/m2 of paclitaxel and carboplatin (AUC 5) every 3 weeks. A follow-up examination conducted 7 years after the fourth surgery showed no evidence of recurrence. |
A 43-year-old premenopausal woman with Gravida 3 and Para 3 came to gynecology outpatient unit with complaints of irregular vaginal bleeding and lower abdomen pain since 6 months. There was no significant medical history. On clinical examination, vital signs were normal, and abdomen was soft with no palpable mass. Abdominal and pelvic ultrasonography (USG) was performed and revealed heterogeneous echogenic mass each in uterine corpus (3.5 cm × 2.5 cm × 2.5 cm) and cervix (1.2 cm × 0.8 cm). A diagnosis of leiomyoma in uterine corpus and cervix with degenerative change was made. Surgical specimen of hysterectomy was received for histopathological examination. Specimen showed a well-circumscribed gray-white to yellow tumor mass (3 cm × 2.5 cm) at upper pole of uterine corpus and yellow tumor mass (1 cm × 1 cm) in cervix [].\nHistopathology examination of the tumor from uterine corpus showed a tumor composed of lobules of mature adipocytes separated by fibrous septae. At places, thin bundles of smooth muscle cells were seen. Small sized normal blood vessels were seen in . There was no nuclear atypia or increased mitotic figures. A diagnosis of lipoleiomyoma was made. Histopathology of tumor from cervix showed a tumor tissue composed of lobules of mature adipocytes, thick fibrous septae [], and few entrapped mucosal glands []. A diagnosis of pure lipoma was made.\nImmunohistochemical (IHC) stains such as smooth muscle actin (SMA) and vimentin were done in sections of both the tumors to confirm the nature of cells. Smooth muscle cells, endothelial cells from the tumor of uterine corpus showed immunoreactivity with SMA and adipocytes were negative [] Vimentin staining showed immunoreactivity with smooth muscle, endothelial cells and mature adipocytes, fibrous septae were negative [].\nIn sections of cervical lipoma, only endothelial cells of blood vessels showed immunoreactivity with SMA, and fibrous septae and mature adipocytes were negative []. Mature adipocytes showed immunopositivity for vimentin []. A diagnosis of lipoleiomyoma in the uterine corpus and pure lipoma in the cervix was confirmed. |
A 58-year-old woman was admitted one year ago to the neurointensive care unit of our hospital for sudden onset of intracerebral and subarachnoid hemorrhage of the right frontal lobe. She had a history of tobacco use and moderate alcohol abuse, hypertension and chronic obstructive pulmonary disease. Her surgical history included left lumpectomy for breast cancer, amputation of the third finger on the right hand and jaw surgery a few years ago. At admission her vital signs were within the normal range: blood pressure 140/87 mmHg, heart rate 71 beats per minute (bpm), respiratory rate 20 breaths per minute and oxygen saturation on ventilator 100%. She was awake and alert but lethargic. The electrocardiogram (ECG) at admission showed normal sinus rhythm (60 bpm) and possible septal necrosis. Right craniotomy for evacuation of the hemorrhage and clipping of right middle cerebral artery aneurysm were performed. Postoperatively, her neurological functions were intact. Troponin I levels showed a slight increase from 0.33 to 3.55 ng/mL during the first two days, and then decreased to 1.96 ng/mL on the third day postsurgery, and similar variations were seen for the levels of creatine kinase-MB fraction (from 4.0 to 11.5 to 4.3 μmol/L). ECG on the second day showed deep negative T waves in almost all leads with prolonged QTc interval []. Due to abnormal myocardial necrosis markers and ECG, a two-dimensional (2D) transthoracic echocardiogram was ordered, and revealed a normal left ventricular cavity size with hyperdynamic basal motion, akinesis of midventricular segments and nearly normal apical motion []. Left ventricular global systolic function was moderately decreased with left ventricular ejection fraction at 35% (normal values >55%). Clinical and echocardiographic features were consistent with stress-induced cardiomyopathy.\nThe next day, the patient underwent coronary angiography, which ruled out any coronary artery disease. However, the ventriculogram surprisingly revealed an aneurysmatic anterior wall [], which then was characterized by another transthoracic echocardiogram as an anteroseptal wall aneurysm with near-normal motion of the other segments []. Global radial strain was calculated and, despite seriously compromised midventricular anteroseptal motion, midventricular-averaged radial strain was at the lower end of normal range (i.e. 16%).\nAs predicted by the global radial strain in the previous examination, another transthoracic echocardiogram performed three weeks later documented the complete resolution of wall motion abnormalities [Figure and ], with completely normal radial strain values []. |
A 33-year-old male renal transplant recipient presented with fatigue, pedal edema, abdominal distension and pain and dragging sensation in the right upper abdomen 4 months after transplantation. He had undergone live related renal transplantation in February 2008 with mother as the donor. His basic kidney disease was presumed to be chronic glomerulonephritis. He was diagnosed with end-stage renal disease in October 2007 and was on maintenance hemodialysis twice a week since then. During his pretransplant evaluation, he was found to have left-sided exudative pleural effusion with high adenosine deaminase level and was started on antitubercular therapy (ATT) with four drugs—rifampicin, isoniazid, ethambutol and pyrazinamide—which he received for four weeks prior to transplantation. He had no clinical evidence of pericarditis during this period and the echocardiogram showed no evidence of pericardial effusion. After renal transplantation the ATT was modified, with rifampicin being replaced with ciprofloxacin as the patient was receiving immunosuppression with tacrolimus. He had a baseline serum creatinine of 1.2 mg/ dl. Physical examination revealed gross bilateral pitting pedal edema, engorged jugular veins in the neck, tense ascites and tender hepatomegaly. He had clinical evidence of bilateral pleural effusion. The heart sounds were heard normally with no murmur or added sounds. Chest X-ray revealed cardiomegaly with a cardiothoracic ratio of 0.6. The pleural fluid and ascitic fluid were transudative in nature. An echocardiogram was performed, which showed a diffusely thickened pericardium and a pericardial effusion of 5 mm posterior to the heart. A computed tomography scan was performed, which showed generalized pericardial thickening with maximum thickness of 7 mm []. No significant pericardial calcifications were noticed. A diagnosis of chronic constrictive pericarditis was made and he underwent anterior pericardiectomy. Histopathological examination of the pericardial tissue did not show any granulomas and the tissue was composed predominantly of fibrous tissue. The edema and ascites subsided and he had good effort tolerance and was discharged with advice to continue ATT. |
A 42-year-old Peruvian man hospitalized in Peru, with recurrent abdominal pain and diagnosis of chronic lithiasic cholecystitis, underwent open cholecystectomy. During the surgical procedure, choledochal cyst and a partial obstruction of the choledochal duct was found. Thus, a hepaticoduodenostomy was performed without removing the cyst. No complications were reported during or after the surgery.\nSeven months after the surgical procedure, he presented with recurrent abdominal pain with belt-like radiation, which was exacerbated with cholecystokinetics, and associated with chronic diarrhea and steatorrhea. He was then referred to our hospital. At admission, his laboratory tests revealed elevated serum amylase (299 U/L) and serum lipase (597 U/L), with remaining liver function tests within normal range. This led to the diagnosis of chronic pancreatitis. During subsequent endoscopic retrograde cholangiopancreatography (ERCP) procedure, pancreas divisum and amputation of the major pancreatic duct (Wirsung) was found (). To confirm the diagnosis, the patient underwent an abdominal computed tomography scan, where a diverticular formation on the pancreatic head was reported. The patient then underwent an exploratory laparotomy, with the operative finding of pancreas divisum, congenital choledochal cyst of 20 mm in diameter and hypoplasia of the distal part of the major pancreatic duct and the major duodenal papilla, which emptied into the hypoplastic portion with flow disruption of the ventral pancreas. With histopathology findings of a major duodenal papilla with chronic inflammation and fibrosis with chronic nonspecific duodenal inflammation, a transduodenal papillectomy was performed. The postoperative course was uneventful.\nThe patient again presented abdominal pain 8 years later, having the same clinical characteristics associated with fever, asthenia, adynamia and involuntary weight loss of 5 kg since the last 3 months. Laboratory studies showed elevated serum amylase and lipase, diagnosing recurrent acute pancreatitis with associated cholangitis. The patient was hospitalized, seven days later he was discharged without complications with normal serum lipase levels, but persistent elevated amylase levels (152 U/L).\nHowever, 7 years later he presented acute cholangitis with following laboratory reports: total bilirubin, direct bilirubin and indirect bilirubin within normal levels, and elevated levels of alkaline phosphatase 171 U/L, aspartate aminotransferase 69 U/L and alanine aminotransferase 67 U/L. The patient then underwent a percutaneous transhepatic cholangiography (PTC), where a hepaticoduodenostomy stenosis was identified. Subsequently, a balloon dilatation and placement of an internal-external biliary catheter was performed. Next, he underwent choledochal cyst resection and dismantling of the hepaticoduodenostomy with Roux-en-Y hepaticojejunostomy, with partial resection of the IV B hepatic segment. The patient was discharged 5 days later, asymptomatic and without any complications.\nThe patient remained asymptomatic for 12 years, after which he presented with acute cholangitis. A percutaneous biliary drainage was performed, revealing a leak to the abdominal cavity. Subsequently, a magnetic resonance imaging showed loss of proximal left branch continuity, a T1-hypointense pericholangitic lesion consistent with inflammatory activity, as well as pneumobilia on the left hepatic lobule, leading to the diagnosis of left hepatic duct stenosis. Subsequently, the patient underwent a hepatojejunostomy of the left duct. Four days after surgery, the patient was discharged with all laboratory levels within normal limits. Currently, the patient remains asymptomatic 18 months after the surgery. |
A 47-year-old postmenopausal woman presented with slowly increasing abdominal girth with a palpable abdominal mass and vaginal bleeding. At the age of 15, she underwent abdominal surgery due to intussusception of the small bowel of unknown cause. During the preoperative evaluation, colonoscopy identified eight hamartomatous polyps, nine hyperplastic polyps, and two tubulovillous adenomas in her large intestine. She had abundant mucocutaneous melanin pigmentation around the lips. Based on her clinical features and the histopathological diagnosis of hamartomatous polyps and mucocutaneous pigmentation, she was clinically diagnosed with PJS. No other family members had clinical signs of PJS. On pelvic and abdominal ultrasonography and computed tomography, she was found to have huge bilateral ovarian cystic masses with ascites in the pelvic cavity. On exploratory laparotomy, the left ovarian mass was ruptured, and approximately 1,500 mL of ascitic fluid was identified. The patient underwent hysterectomy, bilateral salpingo-oophorectomy, and small bowel polypectomy via small bowel enterostomy.\nGrossly, the left ovarian tumor was a multilocular cyst measuring 22×20×6 cm (), whereas the right ovarian tumor was an oligolocular cyst measuring 8×7×5 cm (). The septa of the cysts were thin and the internal surface was smooth. Both cystic tumors contained mucinous fluid without any solid area. Microscopically, both ovarian tumors showed cysts of varying sizes lined by a single layer of columnar epithelium, and the architecture of the glandular arrangement was characterized by clusters of small glands budding from surrounding a centrally located large duct-like structures, forming a lobular arrangement at lower magnification (). The mucinous epithelium had pale eosinophilic cytoplasm with basally located nuclei (). Neither epithelial stratification nor nuclear atypia was identified. There were no foci of sex cord tumor with annular tubules in both ovaries. The uterine cervix, endometrium, and fallopian tubes were meticulously examined, but no histologic evidence of mucinous epithelial lesions or metaplasia was identified.\nPolyps of the small intestine showed a branching polypoid structure with crypts and villi of variable lengths and cystically dilated glands, which were divided by muscularis mucosa branching in various directions (), as highlighted by desmin immunostaining (). On combined Alcian blue (pH 2.5) and periodic acid-Schiff after diastase (DPAS) staining, the intracytoplasmic mucin in the epithelium of the ovarian tumor was negative for Alcian blue pH 2.5 and positive for DPAS. The cytoplasm stained bright pink for combined Alcian blue pH2.5/DPAS, implying that the mucinous contents of the ovarian tumor were neutral (), in contrast to mucin in the normal endocervical mucosa, which was positive for both Alcian blue pH 2.5 and DPAS and showed a purple-violet color for combined Alcian blue pH2.5/DPAS (, inlet).\nWe performed immunohistochemical staining using formalin-fixed, paraffin-embedded, 4-μm-thick tissue sections with an OptiView DAB immunohistochemical detection kit (Roche Diagnostics, Mannheim, Germany) on a Benchmark XT autoimmunostainer (Ventana Medical System, Tucson, AZ, USA). The mucinous epithelium of the ovary showed diffuse immunoreactivity for MUC6 (Novo, Newcastle upon Tyne, UK), moderately intense immunoreactivity for carbonic anhydrase-IX (Novus Biologicals, Littleton, CO, USA), and focal immunoreactivity for MUC5AC (marker of foveolar-type mucin; Novo) and HIK1083 (TOYO 2CHOME, Tokyo, Japan) (–). The epithelium did not show immunoreactivity for p16 (Santa Cruz Biotechnology, Santa Cruz, CA, USA), estrogen receptor (Novo), progesterone receptor (Novo), or p53 (Oncogene, Uniondale, NY, USA). The Ki-67 labelling index was less than 5%.\nGenomic DNA was extracted from peripheral blood samples by a QIAamp DNA blood kit (Qiagen GmbH, Hilden, Germany) and from fresh frozen tissue of the left ovarian tumor by a QuickGene DNA tissue kit (Fujifilm Life Science, Tokyo, Japan). The STK11 gene was amplified via polymerase chain reaction by using 10 sets of primers in intronic flanking regions containing all exons (). Sequencing analysis was performed by a cycle sequencing kit (Thermo Fisher Scientific, Waltham, MA, USA) following the manufacturer’s instructions.\nMultiple ligation-dependent probe amplification (MLPA) was performed to detect deletions and duplications in the STK11 gene by STK11 MLPA kit P101 (MRC-Holland, Amsterdam, The Netherlands). The peak height of the probes was analyzed using GeneMarker software v.1.7 (SoftGenetics LLC, State College, PA, USA). A peak ratio less than 0.65 was interpreted as a ‘deletion,’ whereas a peak ratio greater than 1.35 was interpreted as a ‘duplication.’\nSequencing analysis did not detect any mutation in the exons and exon-intron boundaries of the STK11 gene in the blood and ovarian tumor samples. Using MLPA analyses, however, peripheral blood leukocytes showed a germline heterozygous deletion mutation at exons 1–7 of the STK11 gene (c.1-?_920+?del) and the tumor tissue showed mosaic loss of heterozygosity due to the mutation (heteroplasmy) (–). |
The patient was a 61-year-old man with multiple left rib fractures (1–6 ribs), left pneumothorax, left lung contusion, and left thoracic subcutaneous emphysema due to a fall injury. The examination showed a partial depression in the left front rib and abnormal breathing (see Fig. ).\nAdmission chest CT examination: 1–6 rib fractures on the left side (of which 3, 4 ribs are long comminuted fractures (see Fig. )); left pneumothorax, left traumatic wet lung; a small amount of liquid pneumothorax on the left side.\nPatient was given early chest straps, multiparametric monitoring, analgesia, and oxygen therapy. The chest pain was still severe. The visual analogue scale scored 7–8 points for the pain at rest and 9 points for the cough.\nPhysical examination revealed that the left chest wall was recessed and abnormally breathed. The CT scan of the rib showed a long comminuted fracture of 3 and 4 ribs. The key to successful operation was the reduction and fixation of these two rib fractures. A preoperative CT scan was performed to reconstruct the 3D model based on the scan results (see Fig. ), and 3D printing technology was used to prepare 3 and 4 rib models (see Fig. ). The three D print models of each fracture segment of the two ribs were adherently reconstructed.\nThe two rib metal plates were separately shaped according to the reconstruction model (see Figs. and ).\nThe patient is scheduled to have a open reduction and internal fixation of 3–6 rib fracture. After general anesthesia, right side lying position, small incision about 8 cm was performed under the edge of 4th rib underarm. The skin was sequentially incised and the subcutaneous tissue was freed layer by layer. The front of the latissimus dorsi muscle and the anterior serratus were exposed. The tunnel was established on the 3rd and 4th rib surfaces from the back of the chest and small muscles to the back of the scapula. The special long hooks lifted the scapula and exposed the scapular operation space. With assistance of endoscope, the electrocautery is useful to expose 3 cm outside the outermost fracture lines of the 3 and 4 ribs. The locking plate was molded on the surface of the third rib before operation, and the broken end of the non-fracture at the anterior and posterior portions of the third rib was well fitted. The distance between the two ends of the metal bone plate exceeded the fracture line to 3 nail holes distance. Under the thoracoscope, the metal plate and the ribs were temporarily fixed with long-angled forceps. The MIPO system was used to drill the holes. Two screws were implanted and locked at both ends to firmly fix the metal plate. In turn, each fracture segment was reset and drilled and secured to a metal plate. The fourth rib is fixed in the same way. Intraoperative image (Figs. , and ). 5, 6 rib fractures given to fix the ribs, not the content of this article, not elaborated. Sufficient to stop the bleeding, the wound was given to leave a negative pressure drainage tube. After a routine thoracoscopic probe of the chest cavity, a closed thoracic drainage tube was placed posterior to the 7th intercostal space and the incision was closed layer by layer. After the chest wall is well-shaped. Three days after surgery review the map (Fig. ). |
The patient was an 87-year-old man who had regularly visited another hospital for management of benign prostatic hyperplasia. His medical history included hypertension. In November 2013, a rectal mass was incidentally identified. Colonoscopy showed a large submucosal tumor at the lower rectum (Fig. ). Pathological findings from biopsy specimen showed spindle cells arranged in irregular bands. Mitotic count was 3 per 10 high-power fields, and there was no tumor necrosis. Immunohistochemical staining for h-caldesmon, alfa-SMA, and desmin was positive, while staining for DOG-1, CD117 (c-kit), CD34, and S-100 was negative. Ki-67 index was 40%. The rectal mass was diagnosed as a rectal LMS. The tumor was very large and considered difficult to be removed by surgical intervention in that hospital. However, there were no other effective therapies except for surgical resection. In January 2014, he was therefore referred to our hospital for surgical treatment.\nDigital rectal examination revealed a large mass at the anterior wall of the lower rectum, and the inferior edge of the tumor was located 2 cm from the anal verge. Laboratory findings were within normal ranges. Contrast-enhanced computed tomography (CT) showed that the size of the tumor was 7.5 cm in diameter and that there were no signs of distant metastasis (Fig. ). Abdominal magnetic resonance imaging (MRI) showed that the tumor originated from the anterior wall of the lower rectum and that it was suspected to have invaded the left seminal vesicle and prostate (Fig. ). The tumor was close to the urinary bladder, but a cystoscope showed no signs of tumor invasion. 18F-fluorodeoxyglucose positron-emission tomography showed no distant metastasis.\nWe made a diagnosis of a rectal LMS with suspected invasion of the left seminal vesicle and prostate. In March 2014, we performed an abdominoperineal resection of the rectum combined with both seminal vesicles and part of the prostate. We used the hybrid method of laparoscopic and TaTME approaches for better surgical view because it was apparently difficult to achieve negative surgical margins due to the large tumor located within a narrow pelvic space (Fig. ). In this method, we used five trocars for laparoscopic surgery and a multiple access port for TaTME. A 12-mm trocar was inserted through an umbilical incision, and then the pneumoperitoneum was created. Another 12-mm trocar was placed in the right lower abdominal region, and three 5-mm trocars were inserted in the left lower abdominal and bilateral lateral regions. Pathological findings from the resected specimen showed that the tumor was a pleomorphic LMS and that tumor cells had invaded the left seminal vesicle and prostate with negative surgical margin. No lymph node metastasis was found. Immunohistochemical staining for calponin, alfa-SMA, and desmin was positive, while staining for DOG-1, c-kit, CD34, and S-100 was negative (Fig. ). Tumor necrosis was found in the surgical specimen. The LMS was classified as grade 2 in Federation Nationale des Centres de Lutte le Cancer (FNCLCC) grading and stage IIb in TNM classification. In the postoperative course, the patient suffered from dysuria and needed intermittent self-catheterization. Late-onset urethral injury occurred 30 days after the operation, which was successfully treated with a urethral catheter. The patient was discharged 42 days after the operation. The patient received no adjuvant therapies such as chemotherapy and radiotherapy. Four months later, multiple lung metastases were detected on CT, although local recurrence was not found. The patient died 12 months after the operation.\nIt had been difficult to distinguish gastrointestinal LMS from GIST until c-kit was discovered in 1998 []. That is, the previously diagnosed gastrointestinal LMS is considered to include what is currently termed GIST []. According to the recent literature, the incidence of LMS was reported to be 1/50 to 1/65 of the incidence of GIST [, ]. LMS originates from smooth muscle cells and can arise anywhere in human body, although the main locations of LMS are the retroperitoneum, lower extremities, and the uterus []. Therefore, gastrointestinal LMS is very rare and its characteristics are still unclear.\nTo our knowledge, there are only 10 cases, including our case, in the English-language literature since 1998 in which immunohistochemical findings have been reported [, , ] (Table ). The patients were three men and seven women. Median age was 65 years, ranging from 24 to 88. Average tumor size was 3.8 cm. Polypectomy was performed in two cases, anterior resection in three cases, abdominoperineal resection in four cases, and the surgical method was not reported in one case. Common characteristics of three recurrent cases were non-polypoid appearances such as Borrmann type 2- and plaque-like lesions. Our case had the largest tumor size, and there has been no report in which a large LMS in the lower rectum was removed by the hybrid method of laparoscopic and TaTME approaches.\nFNCLCC grading includes three factors; tumor differentiation, mitotic count, and tumor necrosis []. The Union for International Cancer Control/the American Joint Committee on Cancer staging system for soft tissue sarcoma assesses the following factors; tumor size, tumor depth, lymph node metastasis, distant metastasis, and histological grade []. However, the characteristics of gastrointestinal LMS have not been fully clarified because of its rarity []. Yamamoto et al. [] reported that tumor-specific overall survival rate was 51.6% and that tumor size (≥5 cm) was significantly associated with a poor prognosis. They also suggested that tumor depth and necrotic area might be associated with patient survival. In our case, tumor size and gross appearance could be associated with a poor prognosis, while mitotic count was not. We surmised that patients with at least one risk factor might have a poor prognosis.\nThere has been no standard therapeutic strategy for gastrointestinal LMS. The effect of chemotherapy or radiotherapy is still unclear, whereas some antitumor drugs such as doxorubicin, ifosfamide, and eribulin are suggested to be effective for soft tissue sarcomas [–]. Surgical resection plays a critical role in the treatment for gastrointestinal LMS []. Despite the fact that lymph node metastasis is uncommon in soft tissue sarcoma, lymph node dissection seems to be necessary for gastrointestinal LMS because lymph node metastasis has been reported [].\nIn our case, we performed abdominoperineal resection using the hybrid method of laparoscopic and TaTME approaches. It was considered difficult that the elderly patient underwent a total pelvic exenteration surgery and took care of a urostomy together with a colostomy. We assume that the TaTME approach is useful for the resection of a large pelvic tumor. Surgical margins were negative for tumor cells by this approach, but it was very close (<1 mm). There have been some reports on the effectiveness of radiotherapy against local recurrence [, , ], while radiotherapy was reported to be a risk factor for LMS []. Adjuvant radiotherapy or chemotherapy was not performed because there is no established adjuvant therapy for rectal LMS and the elderly patient did not hope to receive adjuvant therapies. However, local recurrence was not found irrespective of the short surgical margins. The late-onset urethral injury occurred after surgery. We conceived that the self-catheterization might make a negative impact on the occurrence. Four months after the surgery, multiple lung metastases were found. There have been some reports on the efficacy of chemotherapy in other LMS cases [, , , , ]. It also might prevent the recurrence of rectal LMS and contribute to prolong patient survival. |
A 33-year-old woman presented with complaints of shortness of breath, fever, and weight loss for 6 months. On the basis of echocardiography, a diagnosis of ostium primum atrial septal defect (OPASD) with effusive chronic constrictive pericarditis was made. Suspecting tubercular etiology, the patient was started on antitubercular therapy while being worked up for OPASD closure surgery. In the interim, she presented to the emergency department with severe shortness of breath (NYHA class 4) and hypotension. Urgent bedside echocardiography revealed that the effusive constrictive pericardial collection had progressively increased and was beginning to cause symptoms at rest which were unresponsive to medical therapy for almost 3 consecutive months. Therapeutic pericardiocentesis was planned with an aim to relieve the patient of his immediate symptoms and to continue with antituberculous treatment conservatively in the intensive phase to treat the active tubercular disease phase first. A 4.5 French sheath and a J-tipped pigtail catheter were inserted under fluoroscopic guidance in the cardiac catheterization laboratory under monitored anesthesia care. Malposition of the drainage catheter was suspected immediately when the aspirate revealed frank blood. Realizing a cardiac puncture, the sheath-pigtail assembly was secured to the skin with sutures and computed tomography (CT) angiography was urgently done which showed the pigtail catheter piercing the RV free wall and coursing through RV outflow tract and main pulmonary artery [].\nThe patient was subsequently planned for emergency surgical removal of the sheath and pigtail catheter. Intraoperative transesophageal echocardiography (TEE) in the midesophageal RV inflow-outflow view showed the entry point of the sheath and pigtail assembly into the RV anterior free wall []. The pigtail catheter was seen to course toward the RV outflow tract and the main pulmonary artery []. The tricuspid valve above showed moderate tricuspid regurgitation with a RV systolic pressure (RVSP) of 41 mm Hg. The OPASD shunted blood from left to right side []. After sternotomy, the percutaneously inserted sheath and pigtail catheter were seen entering the RV anterior free wall after piercing the thickened pericardium [].\nThe thickened pericardium was gradually dissected off its underlying attachments and excised. After having accomplished systemic heparinization and aortobicaval cannulation, felted stay sutures were obtained around the sheath entry point into the RV and the sheath pigtail assembly was carefully pulled out under TEE guidance, to ensure no further damage has been brought about by it. The cardiac puncture site was directly repaired, and atrial septal defect was subsequently closed on cardiopulmonary bypass. Post bypass TEE revealed mild tricuspid regurgitation with a RVSP of 36 mm Hg. The patient was weaned of cardiopulmonary bypass on dobutamine infusion at 5 mcg/kg/min as mild RV dysfunction was noted, extubated after 4 hours of mechanical ventilation, and discharged on 5th postoperative day. Histopathological evaluation of the pericardial specimen sent for evaluation confirmed tubercular etiology. |
This 2-year-old girl child born to nonconsanguineous parents presented with a history of large head and an abnormal compressible swelling on the right side of the scalp when she was 3 months old []. The swelling increased in size when the child cried. On examination, the child was found to have macrocephaly with delayed milestone in the form of neck holding at 1 year and standing with support at 22 months. On physical examination, there was a large compressible bluish scalp mass on the right parietal region with bony dents underneath. The mass was seen to increase in size on crying. The anterior and posterior fontanelles were open. The child underwent MRI along with MR venography and digital subtraction angiography (DSA) to confirm diagnosis. MRI showed a T1-hypointense swelling in the right parietal region with bony defect communicating with the superior sagittal sinus and ventricular dilatation []. MR venography showed a dilated large extracranial vein on the right parietal region communicating with the superior sagittal sinus through dilated diploic and emissary veins []. MR angiography was normal []. Subsequently, the child underwent DSA under general anesthesia, which confirmed large extracranial scalp vein draining into the superior sagittal sinus seen on the venous phase. Bilateral internal and external carotid arteries were normal. No dural arteriovenous fistula was found []. Thus, the diagnosis of sinus pericranii was made depending on MR and DSA findings. The child was kept on follow-up.\nSinus pericranii is a rare congenital venous anomaly, which is soft and compressible scalp mass that connects directly to the intracranial dural sinuses through dilated diploic and emissary veins. They are known to increase in size on valsalva manoeuvre or raised intracranial pressures and reduce on nondependent positions.[] Mainly congenital, traumatic and spontaneous causes are described for the development of sinus pericranii. Traumatic cause is mainly due to tearing of emissary veins and later developing into communicating blood cyst.[] Pathologically, they can be termed congenital if lined by endothelium.[] Sinus pericranii can appear at any age, usually <30 years, common in males and, although usually asymptomatic, may present with nausea, vomiting and vertigo.[] Differential diagnosis for sinus pericranii includes dural fistula, arteriovenous malformation and other midline masses of scalp-like dermoid, lipoma and encephalocele.[] Sometimes, it can mimic subepicranial varix where there is dilated venous sac on the scalp without communication with intracranial dural sinuses.[] Sinus pericranii may be associated with various other anomalies, like systemic angiomas and craniosynostosis.[] It can be diagnosed by clinical examination and radiological imaging. Usefulness of MR venography and computed tomography angiography has already been described to confidently diagnose sinus pericranii and exclude other mimicking causes of scalp swelling. However, conventional angiography may still be useful to rule out other vascular malformations like dural fistula and arteriovenous malformation. Demonstration of extracranial venous sac communicating with intracranial dural sinus via diploic or emissary veins is necessary for the diagnosis of sinus pericranii, which can be easily performed by today’s imaging modalities.[]\nThe present case showed a large head with developmental delay. Imaging showed mild hydrocephalus. Most of the cases described in the literature are associated with craniosynostosis; however, our case showed macrocephaly. Presence of macrocephaly may be due to hydrocephalus. Developmental delay may be attributed to increased venous pressure in the dural sinuses causing raised intracranial pressures leading to delayed development. Development of transient venous hypertension by sinus pericranii has been described previously.[]\nTreatment options in symptomatic patients include surgical resection or a transvenous endovascular approach. If treatment has to be contemplated, then Gondolfo et al. have recommended to assess the drainage pattern of sinus pericranii. If sinus pericranii is dominant, i.e. if drainage of the brain is through sinus pericranii bypassing the usual venous outlets, then treatment should be avoided.[] However, spontaneous regression of sinus pericranii has also been reported.[] Our case was kept on follow-up under close observation to be treated later if the clinical condition worsened. |
A 45-years-old male engineer with no history of trauma, presented to us with progressive upper back pain for two months that eventually became severe and disabling. The pain was non mechanical and was present even at rest and night. He denied any constitutional symptoms. He had no significant medical history except that he was a smoker for over 20 years.\nOn neurological examination, patient indicated altered sensation from T8 dermatome and below; however, upper and lower limb motor power was normal. He had an unsteady gait and exaggerated deep tendon reflexes in both lower limbs.\nInitial X-rays of the thoracic spine were unremarkable, but MRI demonstrated abnormal marrow replacement and enhancement of the entire T6 vertebra including its posterior elements and right 6th posterior rib. MRI axial cuts at T6 level revealed right postero-lateral epidural extension of the lesion causing severe spinal canal, right lateral recess and right exit foraminal stenosis (, ). In addition, an irregular right lung nodule was noted. CT evaluation confirmed the vertebral involvement () and the presence of a lobulated right lung nodule with emphysematous changes in bilateral upper lobes. Further radiological assessment revealed no other lesion elsewhere.\nDuring the course of the work up, he developed bilateral lower limb weakness and was unable to stand. A diagnosis of thoracic myelopathy due to cord compression was made and immediate surgical management in the form of T6 decompression laminectomy along with T3-T8 posterior instrumentation was performed (). Surgery was uneventful; patient had good recovery and was ambulating independently.\nTissue biopsy from T6 right pedicle revealed presence of metastatic tumour cells with moderate amount of eosinophilic cytoplasm in the marrow spaces suggestive of a metastatic carcinoma with neuro-endocrine features (). Tumour cells were positive for cytokeratin AE1/3, carcinoembrionic antigen (CEA), chromogranin and synaptophysin. Proliferation marker Ki67 was found to be 40%.\nEarly post-operative MRI showed adequate decompression of the spinal canal at T6 level and the patient had interval resolution of symptoms; but there was abnormal fluid collection from the surgical site extending up to the subcutaneous layer, likely to be seroma, which was conservatively managed. Chemotherapy with Carboplatin and Etoposide was initiated after satisfactory wound healing three weeks after surgery.\nOne month after the index surgery, patient developed significant motor deficit in the lower limbs (Right L2-L5 = 2/5; Right S1 = 4/5; Left L2-S1 = 4/5) and had bowel and bladder incontinence. An urgent MRI was done which revealed recurrence of the lesion causing near total obliteration of the spinal canal ().\nA second surgery at this stage was considered high risk and the decision was made to initiate concurrent site specific T4-T7 radiotherapy (30 Gy in 10 fractions) along with chemotherapy. Interestingly at the end of radiotherapy and four courses of chemotherapy (4 months post-op), there was significant clinical improvement of neurology and complete resolution of the soft tissue enhancement surrounding the spinal cord was evident in the MRI (). With appropriate physiotherapy and rehabilitation, patient gradually regained full power in both lower limbs by 6 months. He continues to be under oncology follow up and is ambulant with support. |
A 6-year-old male who presented with complaints of tremors in right upper limb for two months which did not increase in frequency or severity over this period. He had history of occasional holocranial headache for two months. There was no memory or behavioral changes or compromise of school performance. Systemic examination was normal and neurological examination revealed no gross deficit except blurred margins of optic disc. Contrast enhanced CT scan showed large defined isodense mass lesion involving the bithalamic region with continuity in midline. Lateral ventricles were enlarged anteriorly and posteriorly to the mass. There was no enhancement on contrast.\nMR of head showed huge bithalamic lesion, of size about 7 cm × 7 cm × 5 cm interconnected with massa intermedia without any gap in between, occupying whole of the third ventricle, except a part anteriorly. It was uniformly isointense on T1 weighted and, hyperintense on T2 and FLAIR weighted images, with no contrast enhancement.\nThere was no restriction to diffusion weighted images. Extent of lesion was up to midbrain, infiltrating bilateral cerebral peduncles reaching to left medial and posterior temporal and medial occipital lobes. Lateral ventricles were dilated with periventricular lucency and no perilesional edema was present [Figures –]. MR spectroscopy (water suppressed proton MRS) of the tumor revealed choline and creatinine peaks with creatinine peak greater than choline peak. The N acetylaspartate signal was decreased. Small lactate peak was also noticed [].\nAs the mass was very bulky and ventricles were also obstructed, a decision to debulk (and not only biopsy) was taken in order to reduce the mass effect in supratentorial compartment. He was planned for surgery with interhemispheric and transcallosal approach. Lateral ventricles were distorted. A possible tumor decompression was done and a decompression of about 30% was acheived. Intraoperatively tumor was very firm, moderately vascular which was not yielding to suction and to CUSA easily. It was so tough that the excision was probably causing trauma to bilateral thalamic nuclei on account of energetic movement of the hands and instruments. The squash was suggestive of low grade glioma. Considering the total excision impossible, an external ventricular drainage catheter was placed in right ventricle and wound was closed following a good hemostasis. Patient was put on elective ventilation in postoperative period, but it was soon weaned off. The patient deteriorated in sensorium and breathing within 6 h, elective ventilation was again required. Post-operatively the child had a stormy course in the ICU. He remained dependant on the ventilator and continued as unconscious for three weeks. He improved gradually and started opening eyes and was weaned off from the ventilator in further three weeks time. CT scan suggested an operative site cavity and an increase in size of lateral ventricles [Figures and ].\nAs he had high grade fever and CSF culture showed coagulase negative staphalococcus aureus growth, shunt was not done and ommaya reservoir was placed in right frontal region.\nOwing to prolonged need of ventilator, tracheostomy was done. Regular aspiration of CSF was done under aseptic condition for two weeks with coverage of antibiotics according to sensitivity A biventricular shunt was done with y connector with medium pressure chhabra chamber. Patient improved after shunt. Within few days, ventilator was weaned off and decannulation was done. His histopathology showed a moderately cellular tumor composed of mild anisomorphic cells embedded in fibrillary matrix. Tumor cells displayed mild hyperchromasia and pleomorphism. Mitotic figures or endothelial proliferation or necrosis were not seen. Immunohistochemistry revealed tumor cells positive for GFAP. MIB index was low. Synaptophysin positive ganglionic cells were few. KI 67 index was < 2%. Impression was fibrillary astrocytoma (WHO Grade 2). Patient is being discharged on Ryle’s tube and planned for radiotherapy after two months. At the time of discharge, the child was conscious, following commands, breathing spontaneously, tracheostomy site healthy and had started accepting orally. |
The patient was an 18-year-old Spanish male with no relevant medical or psychiatric history. Regarding his substance use, he admitted daily use of tobacco (20 cigarettes/day, meeting criteria for tobacco use disorder), daily use of cannabis (3 cannabis cigarettes/day, meeting criteria for cannabis use disorder), and occasionally weekend drinking (no meeting criteria for alcohol use disorder). He denied consumption of other psychoactive substances. As for his family background there was a paternal uncle with an unspecified chronic mental illness.\nHe was admitted to a psychiatry ward after presenting with an acute psychotic episode. The clinical picture included delusions of reference and persecution, pressured speech, increased alertness, fright, suspiciousness, marked anxiety, and psychomotor agitation during previous days. Hallucinations were not identified. He was aware and oriented in person, place, and time. Over the previous week, he had felt very nervous due to the proximity of high school exams. Thus, he had been drinking about 6 ED cans (80 mg caffeine per can) per day during the last seven days. Also, he had been sleeping less than three hours per night in this period, until finally he showed global insomnia during the day prior to hospital admission. He denied increasing cannabis smoking in these days or consumption of other substances.\nDuring his first day in the inpatient unit he presented severe acute psychomotor agitation that required mechanical restraint and high doses of medication (first olanzapine 20 mg oral, followed by haloperidol 5 mg and levomepromazine 25 mg intramuscularly). After these treatments he exhibited low awareness and the ECG showed sinus bradycardia and QTc prolongation. He stayed 24 hours in the intensive care unit until his ECG was normalized and he recovered good awareness level. Urinary drug screening test was positive for cannabinoids and negative for opioids, benzodiazepines, cocaine, amphetamines, tricyclic antidepressants, and barbiturates. Additional tests (cranial CT and blood tests) were normal.\nThe patient returned to the psychiatry ward and was treated with olanzapine 10 mg daily and there was cessation of caffeinated drinks. After three days, psychotic symptomatology had disappeared and he was discharged with diagnosis of Substance-Induced Psychotic Disorder (according to DSM-5 classification) relative to stimulants (ED) and cannabis. He was followed up at a mental health outpatient setting with positive global evolution. He remained abstinent to cannabis and ED. Antipsychotic treatment was gradually reduced and he was finally discharged of mental health service without pharmacological treatment two years later. |
A 73-year-old man with a history of hepatocellular carcinoma associated with hepatitis C virus-related cirrhosis was referred to the Department of Urology because a thickened wall of the urinary bladder was incidentally detected by computed tomography (CT). The patient had no apparent abdominal and voiding symptoms. He was afebrile, and physical examination findings were almost normal. Routine laboratory examinations were unremarkable; however, urinalysis demonstrated sterile pyuria. Voided urine cytology results demonstrated low-grade urothelial carcinoma. An X-ray film of the kidneys, ureters, and bladder showed no abnormality. CT of the abdomen demonstrated that a linear radiopaque structure, measuring 26 mm in length, traversed the thickened left anterolateral wall of the bladder (). The lesion involved the subserosal part of the sigmoid colon. Cystoscopy revealed a protruding mass on the left anterolateral wall of the bladder, covered with normal-appearing mucosa, with outflow of pus from a shallow recess on the top (). Sigmoidoscopy appeared completely normal.\nThe patient gave a history of ingesting a fish bone accidentally. There may have been a time lag of one month from the ingestion. Clinical history and CT findings strongly suggested an abscess of the bladder wall secondary to migration of a fish bone. Transurethral biopsy of the bladder prior to partial cystectomy was performed because malignancy could not be entirely excluded. When a cold cup biopsy of the mucosa was performed, a linear structure in the submucosa was observed () and removed transurethrally using forceps (). Gross examination revealed one linear, solid, and white-yellow structure, measuring 28 mm in length (). Urinary leak was not detected on cystography, and his Foley catheter was removed on the seventh day after the procedure. We were concerned about complications such as peritonitis or vesicoenteric fistula resulting from the removal of the foreign body, but the postoperative course was uneventful.\nHistopathological findings indicated bladder mucosa with nonspecific chronic inflammation, rich in neutrophils, without evidence of malignancy, and containing putrid skeletal bone, compatible with fish bone. This patient was thought to have a fish bone perforation of the sigmoid colon and subsequent penetration of the bladder.\nAt one month postoperatively, CT scan showed neither a thickened wall of the bladder nor residual fish bone. Urinalysis was normal, and voided urine cytology results were negative. In case, an artifact induced by the fish bone could have led to the false-positive diagnosis of urothelial carcinoma based on the cytology results. |
A 10-year-old castrated male domestic shorthair cat was referred to the Dentistry and Oral Surgery Service of the Matthew J Ryan Veterinary Hospital of the University of Pennsylvania for evaluation of a gingival mass discovered as an incidental finding during a wellness examination 1 month prior to presentation. No obvious signs of oral discomfort were reported. A biopsy performed prior to presentation showed no evidence of malignancy. An odontogenic tumor was suggested at that time.\nNo previous pertinent medical history was reported, and the patient was not receiving any medications at the time. Physical examination, complete blood count and serum biochemistry profile revealed no abnormalities. Given the previous histopathology results, further staging was not pursued.\nAn approximately 1 cm in diameter, raised, erythematous, firm, ulcerated gingival mass was observed on the buccal aspect of the left maxillary third and fourth premolar teeth (). Moderate discomfort was noted on palpation of the mass during the awake oral examination. Gingivitis and calculus accumulation were more evident on these teeth when compared with the contralateral maxillary premolar teeth.\nThe cat underwent general anesthesia for periodontal probing, dental charting, intraoral radiographic assessment and excision of the gingival mass. A left maxillary nerve block was performed for augmented pain control, using 0.4 ml (0.26 mg/kg) bupivacaine (Marcaine; Hospira). Periodontal probing of teeth adjacent to the gingival mass showed moderated build up of dental calculus and gingivitis. Periodontal pockets or malocclusion were not observed. The rest of the oral examination was unremarkable. Dental radiographs showed a well-defined mineralized mass at the alveolar margin overlying the distal aspect of the left maxillary third premolar and the mesial aspect of the maxillary fourth premolar. Some tooth resorption and loss of the periodontal ligament space were noticed ().\nThe oral cavity was rinsed with chlorhexidine gluconate 0.12% solution prior to the mass removal. Then, a rim excision was performed, including 5 mm normal-looking tissue away from the gross and radiographic margins of the mass. A full-thickness incision was made with a #15 scalpel blade. Alveolar and buccal mucosa was raised with a periosteal elevator to expose the underlying bone. Osteotomy of the maxilla was performed with a long #700 carbide bur in a sterile high-speed dental handpiece, taking care not to enter the nasal cavity or the infraorbital canal. The bony tissue was irrigated with sterile saline solution during the osteotomy.\nThe specimen was separated from the maxilla. Remaining root tips of the third and fourth premolar teeth were extracted using a winged dental elevator. Sharp alveolar bone edges were smoothed with a #22 round diamond bur, which was also used to debride the apical area of these sockets. Afterwards the wound was first rinsed with 0.12% chlorhexidine and then rinsed with sterile saline. A buccal flap was sutured to palatal mucosa with 5-0 poliglecaprone 25 (Monocryl; Ethicon) in a simple interrupted pattern (). The cat recovered from the anesthesia uneventfully. Intravenous fluid therapy was maintained for the first 12 h after the surgery, at which time the cat ate soft food and drank water. Postoperatively, amoxicillin-clavulanate (62.5 mg PO q12h for 7 days; Clavamox [Zooetis]) was given orally, while buprenorphine hydrochloride (Buprenex; Reckitt Benckiser) (0.01 mg/kg SL q12h for 5 days) and robenacoxib (Onsior; Novartis) (1 tablet orally q24h for 3 days) were given to provide postoperative analgesia. Chlorhexidine gluconate (Oral Health Tooth Gel; Crosstex) was applied on the oral cavity twice a day for 2 weeks for antiseptic purpose, while soft food was maintained for the same period of time, until the surgical wound healed properly.\nHistopathological examination showed proliferation of mineralized eosinophilic material with multiple, irregularly placed lacunae and reversal lines (), reminiscent of cementum, between the two premolar teeth. The mass was adhered to the distal root of the left maxillary third premolar near the cementoenamel junction, contiguous with and focally replacing the normal cementum layer. The periodontal ligament region was expanded by the mass. The mass extended from the periodontal ligament region into the overlying gingiva, forming a well circumscribed lesion. Histopathology confirmed complete removal of the mass. No recurrence of the tumor was noted by the owner 6 months after surgical excision. |
A 36-year-old White male presented to the emergency department (ED) with a four-month history of left-sided chest pain with associated shortness of breath on exertion. He described the pain as a dull ache that occurred at rest and was not worsened by exertion. He also noted a chronic dry cough that he had for the prior several years that was not associated with illness or exercise and a 12-pound weight loss over the previous month. Past medical history was noted for ureterolithiasis several years prior and he took no medications on a daily basis. His last purified protein derivative skin test was several months prior and was negative. He also denied ever smoking or a family history of autoimmune or inheritable disorders.\nUpon arrival, his vital signs were all within normal limits and his examination exhibited only scant wheezes and coarse breath sounds in the left upper lobe. Egophony and whispered pectoriloquy were both negative in the concerned area. He also had no discernible skin lesions or clubbing.\nElectrocardiogram showed a normal sinus rhythm with 82 beats per minute without any signs of ischemia. Laboratory testing including a complete blood count, complete metabolic profile, and troponin T test were all negative. Chest radiograph (CXR) demonstrated diffuse interstitial nodular opacities throughout the lungs bilaterally with bilateral perihilar consolidations that were worse on the left (). Computed tomography with intravenous contrast of the chest showed mid and upper lung nodularity with a perilymphatic distribution involving the central peribronchial vascular regions as well as subpleural and fissural surfaces causing conglomerate in the upper lobes centrally (). There was also mild symmetric bilateral hilar and mediastinal lymphadenopathy.\nAfter discussion with pulmonology, the differential included lymphoma, tuberculosis, fungal infections, and pulmonary sarcoidosis. The following day, the patient underwent bronchoscopy with endotracheal ultrasound and transbronchial biopsies. Bronchoalveolar lavage was negative for fungal infections, acid-fast bacilli, and malignant cells. Endobronchial biopsies revealed numerous non-necrotizing, well-formed granulomas embedded in dense hyaline sclerosis.\nThe patient was subsequently diagnosed with stage 3 pulmonary sarcoidosis and started on prednisone daily and sulfamethoxazole/trimethoprim three times a week for eight weeks. Following treatment, he had resolution in his symptoms. |
A healthy 8-year-old female child was noted have a growth on her right iris. The asymptomatic lesion was followed for 3 months and demonstrated slight enlargement, leading to suspicion of JXG. She was referred for diagnosis and management.\nOn examination, visual acuity was 20/20 in each eye and intraocular pressures were normal. The only abnormal ocular finding was a minimally pigmented, vascular iris mass, measuring 6.0 mm in diameter and 2 mm in thickness, located in the nasal aspect of the iris []. Gonioscopy and ultrasound biomicroscopy revealed that the lesion was confined to the iris with no extension or seeding into the angle or ciliary body. Fluorescein angiography revealed intense vascularity confined to the mass and not involving the surrounding normal iris []. Our differential diagnosis included amelanotic iris melanoma versus JXG.\nFNAB was performed for diagnostic purposes. Using an operating microscope, a 27-gauge needle was passed through the clear cornea 1 mm from the limbus and directed into the tumor with aspiration using standard technique.[] Cytopathology revealed atypical cells with spindle morphology, ovoid nuclei, and prominent central nucleoli, which were immunoreactive for melanocytic markers melan-A and HMB45, consistent with spindle B malignant melanoma cells [].\nBased on the cytopathological diagnosis of melanoma, basal sector iridectomy was performed. Histopathology revealed an amelanotic neoplasm, composed predominantly of spindle B melanoma cells and few cells with intermediate morphology between spindle B and epithelioid melanoma. There was one mitotic figure per 40 high-power fields []. There was minimal invasion into the trabecular meshwork and base of the pars plicata. The tumor appeared to be completely removed. The tumor was classified as pT2a based on the American Joint Committee on Cancer classification (8th edition).[]\nOne week after surgery, the visual acuity was 20/40, and 6 months later, it remained 20/25. Local and systemic evaluation revealed no evidence of tumor recurrence or metastatic melanoma after 14 years and visual acuity was stable. |
This case involves a 79-year-old female G5P3023 who presented with Clostridium perfringens sepsis. She was in her usual state of health until two days prior to admission to an outside hospital with mental status changes and low back pain. She had fevers and her family reported that she seemed confused and was slow in response. She reported postmenopausal bleeding and lower abdominal pain that began a few days prior to admission. Her past medical history included history of deep venous thrombosis of her left lower extremity on lifelong anticoagulation, hypertension, hyperlipidemia, insulin resistance, hiatal hernia, and early stages of dementia. Her past surgical history was noncontributory. Her obstetrical history was significant for five pregnancies with three vaginal deliveries, a stillbirth, and a spontaneous abortion.\nOn admission to the outside hospital, the patient was febrile to 103°F and white blood cell count (WBC) was 12,500 cells/μL. A urinalysis was suggestive of a urinary tract infection so she was started empirically on vancomycin and ceftriaxone. Initial blood cultures grew gram positive bacilli, so metronidazole was added. CT of the abdomen and pelvis demonstrated an enlarged uterus with endometrial canal thickening and endometrial canal air with surrounding inflammatory stranding, suggestive of endometritis (). Final blood cultures returned on HD3 as Clostridium perfringens, with the source presumed to be endometritis. Her antibiotics were changed to IV ertapenem. Follow-up blood cultures on HD2 and HD3 showed clearance of her bacteremia. She underwent dilation and curettage with an endometrial culture. Pathology revealed fragments of adenocarcinoma in a background of necrosis and the endometrial culture also grew Clostridium perfringens, Bacteroides uniformis, and scant Peptoniphilus asaccharolyticus. The patient was clinically stable and was transferred to our tertiary care center for management of her adenocarcinoma in the setting of Clostridium perfringens endometritis and septicemia after seven days at the outside hospital and seven days on IV antibiotics. Over time at the outside institution, her mental status improved and was clinically stable.\nOn evaluation after transfer, the patient felt well and denied pain or nausea. She was tolerating oral intake, voiding, having regular bowel movements, and ambulating. On exam, her vitals were normal and her BMI was 28.68 kg/m2. Her abdomen was soft, nondistended, and nontender with no rebound or guarding. Her extremities were warm and well perfused with significant venous stasis changes on bilateral lower extremities with associated skin breakdown. Her neurological exam was grossly intact. On pelvic exam, she had a smooth cervix and small 6-week sized mobile uterus, and no adnexal masses were appreciated. No purulent discharge was noted.\nOn admission to our institution (HD 7), laboratory values were significant for normal electrolytes, INR of 3.4, and white blood cell count of 10,000 cells/μL. The IV ertapenem was continued. CT scan of the chest, abdomen, and pelvis was repeated, which showed a decrease in the gas component compared to the previous exam seven days earlier. The patient was clinically stable so preoperative planning was undertaken and included reversal of therapeutic anticoagulation. She received vitamin K on HD 7 with a repeat INR of 2.1. She was then given two units of fresh frozen plasma on HD 8 prior to surgery.\nOn HD 8, she underwent a total laparoscopic hysterectomy and bilateral salpingo-oophorectomy. Given her recent clinical condition, decision was made prior to surgery to forego lymphadenectomy which was discussed with patient. Intraoperative findings revealed purulence draining from the cervical os. The uterus was enlarged and globular, but mobile. Her fallopian tubes and ovaries were normal in appearance. There were no adnexal masses or evidence of metastatic disease. The procedure was uncomplicated and estimated blood loss was 300 cc. Intraoperatively, she received one unit of packed red blood cells due to acute blood loss superimposed on chronic anemia. Her preoperative hemoglobin was 8.2. After the procedure, she was transitioned from IV ertapenem to IV ceftriaxone and oral clindamycin per infectious disease consultant recommendations to provide excellent coverage for clostridial bacteremia and enteric gram negative rods in the immediate postoperative setting. Postoperative course was uncomplicated and she was discharged to a skilled nursing facility on HD 10/POD 2. She was discharged on oral ciprofloxacin with a plan for three additional days of treatment and oral clindamycin for fourteen additional days.\nA picture of the bisected gross specimen is shown in . The cervix is directly over the 8-9 cm marker of the ruler; the left tube and ovary are clearly visualized with the right tube visible at approximately 10 o'clock behind the necrotic intrauterine mass. Pathology revealed an undifferentiated uterine sarcoma mainly in the endometrium with invasion into the inner half of the myometrium. On gross examination, the endometrial cavity revealed an exophytic and friable mass (6.2 × 5.4 × 2.3 cm) located 2.2 cm from the lower uterine segment. Additional detached fragments of loose pink-tank, friable tumor mass (5.5 × 5.0 × 2.0 cm) were also submitted for histologic examination. The cervix, bilateral ovaries, and fallopian tubes were not grossly involved.\nMicroscopically, the tumor was centered in the endometrium and was composed entirely of spindled to oval cells (Figures –). Mitoses were readily identified (, circles). There were large areas of necrosis. Differential diagnostic considerations included carcinosarcoma (malignant mixed Müllerian tumor/MMMT), endometrial stromal sarcoma (both low and high grade variants), adenosarcoma with sarcomatous overgrowth, and leiomyosarcoma. While there were a few mildly atypical glandular epithelial elements, no overtly carcinomatous component was identified, militating against the diagnosis of carcinosarcoma (Figures and ).\nImmunohistochemically, the malignant cells demonstrated patchy positivity for cytokeratin AE1/AE3, ER (estrogen receptor), PR (progesterone receptor), CD10, cyclin D1, h-caldesmon, desmin, and SMA (smooth muscle actin) and were negative for myogenin, ALK, and DOG-1 (not shown). Fluorescence in situ hybridization (FISH) testing for JAZF1, PHF1, and YWHAE rearrangements was negative.\nThe pathology slides of the dilation and curettage from the outside hospital were reviewed and compared to the hysterectomy specimen. While specimens did contain some mildly atypical glandular epithelial elements, they were detached and did not have an overtly malignant morphology. When evaluated in the context of the hysterectomy specimen, which contained no carcinomatous component, we interpreted that glandular component as fragments of benign cervix/endometrium.\nThe patient was followed up in the office 5 weeks after surgery and had recovered well although still requiring intermittent nursing care at home. Adjuvant chemotherapy with gemcitabine and docetaxel was recommended at that time and arrangements were made to see a medical oncologist closer to her home. Ultimately, she was diagnosed with a second primary lung cancer a short time after her recovery which was not apparent on her imaging for this hospital admission. A few months later she died from what was thought to be diffuse metastatic disease with multiple lung masses by the physicians caring for her at that time. |
A 50-year-old female repeatedly presented to our hospital with a cough for 3 months. No other symptoms were associated with the cough. The findings during the physical examination were unremarkable except that her Body Mass Index was 28.5. Contrast-enhanced computed tomography imaging of her chest revealed a posterior superior mediastinal lesion with an area of approximately 2.7 × 1.3 cm () and a value of approximately 122 HU. The mediastinal lesion manifested as an intensely enhancing mass with a circumscribed and well-defined margin. A bronchoscopy showed that the membranous trachea was raised, a mass oppressing the membranous region of the trachea from the posterior mediastinum. No pedunculated or polypoid tumor was in the trachea lumen.\nA surgical excision was performed using a right thoracoscopy. This procedure revealed a well-circumscribed vascular lesion that was located above the carina and behind the trachea that measured 1.5 cm in length and was highly vascularized. Additionally, the tumor closely adhered to the membranes of the trachea. During the process of tumor resection, to decrease tension, we emptied the balloon of an endotracheal tube with the assistance of anesthetist. After the lesion was resected, the endotracheal tube cuff was inflated and extruded into the chest. Regretfully, we ruptured the membranes in the trachea, which resulted in a longitudinal tear that was approximately 0.8 cm in length. This tear was sutured with interrupted stitches using 4-0 Prolene sutures and a pad of pleura. It was further reinforced with a pedicled pleural flap to avoid small amounts of air from leaking from the suture line (). There was no air leakage when an airway inflation pressure of 20 cm of H2O was applied. The patient experienced an uneventful recovery and no postoperative complications and was discharged home on the 16th postoperative day. Fiberoptic bronchoscopy was performed 1 week, 3 months after surgery. No incidence of tracheostenosis occurred.\nOn gross examination, the specimen was a relatively well-demarcated solid mass that measured 2.7 × 1.5 cm. The surface of a section of the tissue was reddish-grey and pliable in texture. Microscopically, the neoplastic cells contained a moderate amount of cytoplasm and small and round-oval nuclei, and a few mitotic structures unsystematically surrounded the vessel. Immunohistochemistry demonstrated that the cells were positive for SMA (smooth muscle actin) and h-cald and negative for CD34, Desmin, HMB45, CK, Syn, and S100 (). The tumor was diagnosed as a mediastinal glomus tumor according to its histological and immunophenotypic characteristics. |
We report the case of a 44-year-old female presenting to the General Surgery clinic with subcutaneous epigastric swelling for 2 years. The patient reported progressive growth for the 3 months preceding the consultation, becoming mildly painful during that period. There were no other accompanying symptoms, namely change in bowel habits or weight loss.\nOn physical examination, there was a 5 cm epigastric mass, corresponding to an epigastric hernia with a 15 mm neck. It protruded with Valsalva maneuver, but it did not reduce while lying supine. The apparently fatty contents of the hernia were incarcerated, but painless. Ultrasound confirmed the presence of epigastric hernia. Blood tests were unremarkable.\nThe patient was admitted for laparoscopic hernia repair, however, during port introduction, there was leakage of moderate amount of mucoid, bright yellow peritoneal fluid. On inspection, we found abundant mucinous ascites and gelatinous deposits covering most of the intra-abdominal organs and omentum and part of the abdominal wall (Fig. A), particularly in the pelvic recesses. The hernia sac was comprised of gelatinous implants (Fig. B). The appendix had its tip engorged by mucinous material (Fig. C). The right ovary was apparently involved by the same mucinous tumor as the appendix (Fig. D). We estimated a peritoneal cancer index of 24. The lack of intestine in the hernia and the operative findings compatible with PMP led to the decision of postponing hernia repair until definitive diagnosis was established. Peritoneal fluid and omentum samples were taken for cyto-histopathology.\nCytology found no malignant cells in the peritoneal fluid. Histology subsequently reported peritoneum extensively covered by mucus and bundles of cylindrical and caliciform epithelial cells. The tumor cells stained positively for CK20, CDX2 and negatively for CK7, favoring a primary bowel malignancy.\nThe patient was discharged on the day following surgery and posteriorly sent to a referral unit for peritoneal surface malignancies, where she underwent cytoreductive surgery and hyperthermic intraperitoneal chemotherapy (HIPEC). |
A 7 year-old girl was admitted to the Neurology Department with severe headache, vomiting, muscle weakness and balance disorders. She had no history of previous disease and her psychomotor development was normal. However, 2–3 months before admission, the child’s behaviour showed problems with concentration. Preoperative computed tomography (CT) revealed the existence of a large cystic tumour with peripheral calcifications expanding from the sella to the third ventricle area and acute hydrocephalus (Fig. ).\nMagnetic resonance imaging identified a large solid pathological area with fluid accumulation in the sellar and suprasellar region (Fig. ).\nBased on these findings, the patient was diagnosed with a tumour expanding from the sella to the third ventricle, most likely an adamantinomatous craniopharyngioma with concomitant acute occlusive hydrocephalus. The recommended treatment included a right-sided pterional craniotomy with extensive resection of the tumour.\nA deterioration in the patent’s status necessitated immediate surgery with resection of the tumour. A pterional craniotomy was performed on the right side under general anaesthesia with the patient in a supine position. After reaching the base of the brain and evacuation of fluid from basal cisterns, the tumour’s wall was cut, and a thick yellowish fluid was drained. The tumour was separated from the right optic nerve, right ICA, optic chiasm and left optic nerve. Instead of the pituitary stalk, two thin bands were found, extending from the third ventricle and running towards the sella turcica on both sides of the tumour. After separating the tumour from those bands, it was incised slightly above the sellar diaphragm, dissected from the third ventricle wall, along the lamina terminalis, and removed. After achieving haemostasis, the dura mater was sutured and fixed to the bone and a cranioplasty was performed. A suction drain was then placed, soft tissues were closed in layers and protected with a dressing. Pathomorphological examination of the surgical specimen revealed a craniopharyngioma G1-WHO testing positively for AE1/AE2 (stratified epithelium), GFAP (reactive gliosis), MIB-1 (with a few epithelial and inflammatory cells) and CD 68 (multiple macrophages, including polynuclear ones).\nThe girl was stable, conscious with good intervention and showed no paresis. She received prophylactic vancomycin, meropenem and fluconazole, fluids for improving water-electrolyte balance, substitution therapy with thyroid and adrenal hormones, and the treatment of diabetes insipidus. The first 6 days after the surgery were without complications. Neurological examination showed no signs of focal damage to the central nervous system. Due to multi-hormonal pituitary insufficiency, substitution therapy was introduced with L-thyroxine, hydrocortisone and desmopressin, adjusting the doses to serum endogenous hormones.\nOn the seventh day after surgery, the patient’s status rapidly deteriorated. She reported a severe headache and had a fever. The patient became sleepy and apathetic. She periodically loss consciousness and had recurrent seizures lasting approximately 15 s. A neurological examination revealed stiffening of the neck, positive Kernig’s and Babinski signs on the right side, and convergent strabismus in the left eye.\nThe result of a head CT did not suggest the need for repeat surgery. A lumbar puncture was performed. CSF was fully transparent with 18 cells per μl (normal range: 0–5), 60% of neutrophils and 38% of lymphocytes, a positive Pandy’s reaction, a negative Nonne-Apelt reaction, 154.0 mg/dl of protein (normal range: 15–45), a normal concentration of glucose (79 mg%, normal range: 50–80), 2.7 mmol/l of lactic acid (normal range: 0.5–2.2). A latex test and aerobic and anaerobic cultures of CSF yielded negative results. The patient tested negatively for anti-CMV, anti-EBV, anti-HSV, anti-poxvirus and anti-Borrelia burgdorferi IgM antibodies. The results of other tests, including aerobic and anaerobic cultures of the blood (collected at peak fever) and urine, were also negative. A microbiological examination of throat and nasal swabs revealed a presence of physiological microflora: Neisseria spp. and Streptococcus viridans.\nBecause of a lack of an etiological factor for bacterial or viral meningitis, the patient was diagnosed with aseptic (chemical) meningitis. Antibiotic therapy treatment included vancomycin, ceftriaxone, fluconazole, acyclovir, and nystatin and was maintained for 14 days. An antiepileptic agent, valproic acid, was administered since both clinical presentation and ictal electroencephalographic findings suggested epilepsy. Hormonal treatment was continued. The patient was fed enterally via a catheter with a semiliquid low-sodium diet.\nIncreasing intracranial pressure required the placement of external drainage on day 11 after tumour resection. On day 14 post-surgery, the external drainage was removed following a CT scan of the head which showed the ventricular system without features of hydrocephalus. After removal of the drain, the patient was in good general status and was verbally responsive. Neurological examination showed the presence of meningeal symptoms and slight strabismus in the left eye, decreased muscle tone and diminished asymmetrical tendineae reflexes in the upper extremities, with a predominance of the left side, and a decrease in muscle tone with areflexia and a positive Babinski sign on the right side in the lower extremities.\nA follow-up MRI scan was carried out on day 152 after surgery. The images were compared with those obtained before treatment. Turbo spin echo diffusion-weighted images and dark fluid sequences with T1- and T2-weighted images in sagittal, coronal and transverse views were obtained prior to and after intravenous injection of a paramagnetic. Status after surgical resection of the craniopharyngioma with possible small remnant of solid part of the tumour in sella is represented in Fig. .\nThe patient underwent systematic rehabilitation and monitoring of the treatment for multi-hormonal pituitary insufficiency. Six months after the procedure she still needed substitutional hormone therapy, anti-epileptic treatment and ophthalmic and psychological counselling. A systematic improvement in the patient’s condition was noticeable. Currently, the patient is in good neurological condition without focal damage to the central nervous system. Hormone substitution continues. |
An 88-year-old man had presented with a sudden onset of severe hoarseness of his voice. The more common causes for his symptoms had been ruled out by pulmonology medicine and video laryngoscopy, which showed left vocal cord paralysis, before our examination. The patient's surgical history included uneventful staged bilateral carotid endarterectomy in 2002. Thoracic computed tomography revealed a 4.3 × 5.2 × 5.0 cm saccular thoracic aneurysm in zones 1 through 3 of his aorta (, A and B). Because of the patient's age and multiple comorbidities, including calcific plaque in and near the ostium of all three brachiocephalic vessels, open debranching was not considered; rather, a staged hybrid repair was selected.\nThe initial procedure was a right-to-left carotid–carotid bypass performed via a retroesophageal route using a 6-mm ringed polytetrafluoroethylene graft. This was followed by ligation of the proximal left common carotid artery. Scarring from the patient's previous cervical interventions and calcification of the proximal common carotid arteries added to the technical challenge of the procedure. A left carotid–left subclavian bypass was not performed in the same setting because of further scarring at the left supraclavicular area; thus, revascularization of the left arm was planned for the endovascular portion in the subsequent procedure. Postoperatively, the patient showed no signs of a neurologic deficit apart from hoarseness that continued from his preoperative condition.\nThe second procedure was initiated by direct cutdown and cannulation of the right femoral artery and left brachial artery, percutaneous access to the left femoral artery, and right radial artery access for placement of a marker wire in the brachiocephalic artery. A 38-mm covered stent-graft was deployed just before the origin of the innominate artery to cover the origin of the left carotid and left subclavian arteries. A second 34-mm covered stent-graft was placed distally. Type Ia and type II endoleaks were observed with initial angiography, the latter originating from retrograde flow via the left vertebral artery (). The proximal landing zone of the covered stent-grafts was ballooned again, successfully eliminating the type Ia endoleak. Laser-directed graft fenestration at the left subclavian artery could not be performed because perpendicular alignment with the graft could not be achieved. The procedure was, therefore, terminated with a type II endoleak, which significantly decreased after heparin reversal. Embolization and extra-anatomic bypass would be considered, if required, if left upper extremity symptoms or a persistent leak developed. The patient was extubated without new-onset peripheral or neurologic ischemic symptoms.\nAt the 2-week follow-up visit, satisfactory perfusion of the left upper extremity was present, with a nonpalpable left radial pulse, the trunk of the left subclavian artery was thrombosed, and left vertebral flow remained, as expected, reversed. For educational and anatomic reference, an artist's depiction of the left recurrent laryngeal nerve location and surrounding anatomy is shown in . The patient provided written informed consent for the report of his case and imaging studies. |
A 74-year-old male patient underwent left total knee arthroplasty (TKA) at our institution for degenerative knee osteoarthrosis. He had an uneventful postoperative course with no history of delayed wound healing or persistent drainage. Four years later, he presented with a 2-month history of a gradually developing painless swelling over the anterior aspect of the operated knee; the swelling was associated with a small sinus that was extruding a straw-coloured fluid. He had no history of fever, decreased appetite, or weight loss. He had no other musculoskeletal, respiratory, or systemic symptoms of note. He had no history of antecedent trauma, recent travel, or contact with infectious diseases. The patient is a known hypertensive, but the blood pressure was well controlled with treatment, and he is otherwise healthy. He is a retired teacher with no history of involvement in activities requiring excessive kneeling. He is ambulatory in his community and can walk comfortably with the assistance of a cane.\nThe patient's general physical examination results were within normal limits; positive physical findings were limited to the involved knee. There was an anterior knee swelling involving mainly the prepatellar area, approximately 7 cm in diameter, fluctuant, and not tender to palpation, with minimal surrounding erythema; the erythema was present mainly at the punctum. The punctum was draining a yellowish discharge on pressure (). There was no bony tenderness at the patella, distal femur, or proximal tibia. There was no detectable knee effusion, instability, or crepitus. The range of motion was well preserved (5–110°), as it was a prosthetic knee. It was only painful at the end of flexion as this movement compressed the prepatellar bursa.\nPlain radiographs of the knee showed a prepatellar soft tissue swelling (). There were no obvious bony changes, osteolysis, or loosening at the bone-prosthesis interface. Needle aspiration of the prepatellar bursa yielded 50 mL of slightly turbid straw-coloured yellowish fluid that was sent as per our protocol for cell count determination, microcrystal analysis, Gram staining, and Ziehl–Neelsen (ZN) staining for acid-fast bacilli (). Cultures for aerobic and anaerobic bacteria, Mycobacterium tuberculosis, Brucella, and fungi were requested.\nConsidering the patient's age, chronic presentation, and the presence of sinus, he was admitted with a diagnosis of infected prepatellar bursitis. The primary aims of admission were wound dressing, awaiting aspiration results, and further work-up. Aspiration results revealed normal cell counts, no crystals, and no organisms on Gram stain. To our surprise, ZN stain revealed acid-fast bacilli consistent with typical tuberculous infection; the bacterium was confirmed on culture 6 weeks later as Mycobacterium tuberculosis-sensitive to rifampicin, isoniazid, ethambutol, and streptomycin. Cultures for bacteria, Brucella, and fungi were negative. Aspiration was repeated and yielded similar results. The blood work-up showed normal total and differential white blood cell count and slight elevation of both erythrocyte sedimentation rate (ESR) (80 mm/hr; normal: 30–70 mm/hr) and C-reactive protein (CRP) (8 mg/L; normal: 0–4 mg/L). Tuberculin skin test revealed a negative result (<5 mm induration at 72 hrs), and further work-up including chest radiograph and echocardiogram revealed no evidence of systemic disease. A triple three-phase bone scan displayed normal uptake both at the bone-prosthesis interface and at the patella. An infectious disease consult was obtained. The patient was started on rifampicin (600 mg/day), isoniazid (300 mg/day), pyrazinamide (1500 mg/day), and ethambutol (800 mg/day) for 2 months and continued on the same dosage of rifampicin and isoniazid to complete a 6-month course.\nThree weeks later, the swelling significantly subsided in size and the sinus healed; therefore, the patient was discharged. He was reviewed at 6-week intervals at both the orthopaedic and infectious disease clinics for clinical progression and for any side effects of the medication. At the most recent 6-year follow-up, he was doing well with no evidence of local recurrence or prosthetic loosening (). |
A forty-year old Caucasian male was referred by his general practitioner for urological assessment because of persisting, painless hemospermia, which had initially been treated with several courses of oral antibiotics for almost seven months. The patient denied prior trauma or genitourinary tract infection. The patient denied a past medical history of bleeding disorders, hypertension or malignancy of the urinary tract. On physical examination there were no signs of malignancy except a moderate swelling of the right inguinal lymph nodes. External genitalia, the prostate on digital rectal examination, the vasa deferentia, abdomen and pulmo were found to be normal. Urinalysis was sterile with a moderate microscopic hematuria of 25 Erythrocytes/μl (normally absent in urine). Full Blood Count, prostate specific antigen (PSA) level and clotting was normal. Semen analysis for the presence of melanospermia was not performed. Transrectal ultrasound (TRUS) showed a normal prostate of 35 cm3 and a round, hypo echoic, solid mass measuring 19 × 16 mm within the right seminal vesicle (Figure ). The left seminal vesicle appeared normal. The lesion was further investigated by magnetic resonance imaging (MRI) of the pelvis confirming the ultrasound finding (Figure ).\nIn order to assess the nature of this suspicious lesion the patient underwent transrectal ultrasound (TRUS) guided biopsy of the right seminal vesicle. Histopathology revealed infiltration of the right seminal vesicle with strongly pigmented melanoma cells positive for S 100 and Hematoxylin-Eosin representing metastatic disease and was negative for Cytokeratines AE1/3 (Figures and ).\nS 100 is a sensitive immunohistochemical marker for melanoma and belongs to the family of calcium binding proteins such as calmodulin and troponin C. It is localized in the cytoplasma and the nucleus of the cell and is divided in two subunits namely S100A and S100B. Although highly sensitive for melanomas and their metastases, is also expressed in non-melanocytic tumours such as Schwannommas, glial and neural cells and Langerhans cells.\nHematoxylin-Eosin staining on the other hand is a typical histological staining and not an immunohistochemical marker as S100. It has been in use for more than a century and is still an essential tool for recognizing various tissue types. Hematoxylin has a blue-purple color and stains nucleic acids. Eosin is pink in color and stains proteins nonspecifically. Hematoxylin-Eosin staining is also used for the measurement of tumour thickness, which is also known as the Breslow thickness, and represents a main prognostic factor for melanoma.\nIn our try to locate the primary site of the melanoma, dermatological assessment and computed tomography (CT) of both thorax and abdomen were performed. While pulmonary, hepatic and supraclavicular lymph node metastasis were found, the primary site of the melanoma remained unidentified. Since no primary site of the melanoma could be identified a melanoma of unknown primary was finally diagnosed.\nThe case entered our institutional multidisciplinary tumor board. Following respective recommendation the patient was included into an adequate clinical trial (EORTC 18032 Study) [].\nDespite active treatments in both arms the disease remained progressive. Following a further institutional multidisciplinary tumor board consensus, the supraclavicular lymph nodes and pulmonary masses were resected revealing largely necrotic metastases of the malignant melanoma. Subsequently further progression occurred and the patient refused any further specific therapy. He succumbed six months later to his progressive disease. |
A 47-year-old male Han Chinese motor-repair worker was admitted to the department of ophthalmology of our hospital due to massive hemorrhage in his left eye after he bit an automobile tire tube at 12:00 a.m. on February 9, 2011. The physical examination revealed extensive rupture of the tarsal plate of his left upper eyelid, and lacrimal canaliculus rupture and bulbar conjunctival hemorrhage were observed. A computed tomography (CT) scan indicated subarachnoid hemorrhage. No abnormality was observed after abdominal ultrasound examination. A chest X-ray indicated pulmonary contusion (60% of the total volume).\nClosed drainage of his right pleural cavity was performed via the second intercostal space at 4:00 p.m. on February 10, 2011. Sudden chest pain was reported by the patient 12 hours later. A physical examination showed that his heart rate was 120 beats per minute, his temperature was 38°C, his blood pressure was normal, and his respiratory rate was 25 breaths per minute. Chest ultrasound detection was then performed, which revealed severe pleural effusion in his right thoracic cavity. Subsequently, closed drainage of the pleural cavity was performed via the seventh intercostal space, and approximately 800mL of yellow hydrothorax was extracted. A chest CT and barium swallow examination showed an irregular tear in the middle and lower portions of his esophagus (Figure ). The patient was then transferred to our department of thoracic surgery immediately. For the treatment, drainage of empyema (right chest), removal of thoracic esophagus, exposure of cervical esophagus, cardiac ligation and gastrostomy were performed respectively. Severe inflammation of his right thoracic cavity was observed during the operation. There was a large amount of purulent secretion (approximately 1500mL) in his right thoracic cavity. As exposure of cervical esophagus was noticed during the operation, an ostomy bag (ConvaTec Shanghai Ltd., Shanghai, China) was used to collect the secretions (e.g. saliva) from his esophagus. Meanwhile, gastrostomy feeding and therapeutic antibiotics (10 days) were given. The patient could drink water 72 hours later. The water could directly flow to the ostomy bag. The patient was discharged 10 days later. A successful esophagogastrostomy was obtained 3 months after the first surgery using a gastric tube (4cm in width) constructed from the greater gastric curvature. |
A 49-year-old woman visited the emergency room for acute chest pain radiating to the back. She had known hypertension: the blood pressure in the left upper limb, right upper limb, left lower limb, and right lower limb was 210/65, 218/60, 176/90, and 175/90 mm Hg, respectively. The D-dimer level was normal at initial admission. An electrocardiogram showed a normal sinus rhythm. Considering that the patient had typical clinical symptoms of acute chest pain radiating to the back, with asymmetry of limb blood pressures, and a normal D-dimer level and electrocardiogram, the diagnosis of IMH was suspected. Chest nonenhanced computed tomography (CT) was performed, which showed crescent-shaped thickening of the thoracic aorta wall with the same attenuation as that of the lumen and linear calcification ingression (). Computed tomography angiography (CTA) revealed an intimal flap and expanded false lumen with slight enhancement (). Digital subtraction angiography showed mild stenosis in the thoracic aortic lesion without an obvious intimal flap (). Based on both the clinical symptoms and diagnostic test results, only thoracic IMH was considered by both radiologists and cardiothoracic surgeons; hence, thoracic endovascular aortic repair and not surgery was considered at that time. Then, an endovascular stent (30 mm × 200 mm, Medtronic) was implanted into the patient. Completion angiography demonstrated that the thoracic aorta and branches were patent, with no obvious endoleak or extravasation (). The prevalence of acute chest pain radiating to the back decreased after treatment, and the patient recovered well and was discharged home.\nThree months after endovascular stent graft implantation, follow-up CTA () showed enlargement of the false lumen with nonhomogeneous enhancement and mediastinal extensions. The patient was interpreted by radiologists as having an endoleak and periaortic/mediastinal hematoma, which were also considered by other medical centers during follow-up. However, seven months later, the patient was readmitted to the hospital with complaints of dysphagia and dyspnea. Subsequent magnetic resonance imaging () showed gradual enlargement of the false lumen with mediastinal extensions and new lesions in the azygos vein. Angiogenic hemangioma was suspected, and endobronchial ultrasound biopsy was performed to confirm the lesion. Histopathological evaluation indicated an intimal sarcoma (). Immunohistochemical staining for vimentin, CD10, CD68, and CD99 was positive, whereas that for S-100, CD56, desmin, CD30, CD15, leukocyte common antigen, smooth muscle actin, CD34, CD61, and CD5 was negative. Afterwards, she received adjuvant chemotherapy with a combined treatment of ifosfamide and epirubicin; however, the general condition of the patient rapidly deteriorated after 1 cycle of adjuvant chemoradiotherapy, and she died exactly 19 months after the initial hospitalization. |
A 28-year-old female with insignificant past medico surgical history presented with one day of acute onset pain in the periumbilical region that later migrated and confined to the RIF. She had associated intermittent fever, nausea, and loss of appetite. She did not have any urinary symptoms, bowel irregularities, or gynecological complaints. Abdominal examination was performed by two senior surgeons at two different occasions; the same day had findings of guarding and rebound tenderness at RIF. Hematological tests showed polymorphonuclear leukocytosis with left shift. Biochemical tests and urinalysis were normal. Urinary pregnancy test was negative. Abdominal radiographs were unremarkable. USG could not visualize appendix and was inconclusive except for probe tenderness in RIF. CT scan of the abdomen could not be done due to unavailability. A clinical diagnosis of acute appendicitis was made assigning an Alvarado score of 9/10. Laparotomy was performed using the Lanz incision in RIF. Intraoperatively appendix was found to be normal without evidence of inflammation or infection in RIF. In view of symptoms and signs, a possibility of other pathology was thought. Walking the bowel proximally up to 3 feet (1 m) did not show a Meckel's diverticulum or any other small bowel lesions. There were no obvious mesenteric lymph nodal enlargement and pelvic organs looked pristine. Approaching closure, just when the medial edge of the incision was retracted superomedially, a hemorrhagic lesion seemed to appear little deeper in the mid abdomen. Therefore, the incision was extended transversely from the medial edge to explore further. Entire bowel was explored and this revealed an ulcerated lesion measuring 7 × 5 cm arising from the antimesenteric border of the ileum 8 feet (2.5 m) from ICJ with localized interloop hemoperitoneum and inflammatory exudates as shown in . Resection of ileal segment containing the lesion was performed followed by restoration of bowel continuity and peritoneal toileting. The lesion was subsequently reported to be an ulcerated malignant ileal GIST.\nHistopathologically, gross examination confirmed the operative findings, and the cut section revealed a nodular lesion protruding out of the serosal surface measuring 7 × 5 cm along with 2 lymph nodes each measuring 2 × 1 cm.\nMicroscopically, the growth from the ileum had villous lining epithelium with focal ulceration. The submucosal region had a circumscribed nodule with proliferation of loosely cohesive spindle cells; some of which were arranged in vague storiform pattern and others in long fascicles. There were areas with epitheloid cells forming small anastomosing nests and cords. The areas in between these showed skenoid fibers along with focal areas of hemorrhage, infarction, and congestion as shown in . The mitotic figures were seen (8/50 high-power field). The lymph nodes were microscopically identified to be reactive, and the resected margins of the ileum were free of tumor.\nBased on tumor size and mitotic activity, possibility of a malignant GIST was suggested along with immunohistochemical analysis (CD117 and CD34) for further confirmation. The patient had an uneventful recovery and was discharged on the 8th postoperative day. She was advised to review a week later at the outpatients but failed to report. All possible contacts were used to trace her, but she remained inaccessible and lost to follow-up. |
Sixty-three-year-old woman presented with multicentric left breast cancer requiring mastectomy. She is 69 inches tall with a weight of 215 pounds and a corresponding BMI of 31.8 kg/m2. She has a history of inferior pedicle breast reduction surgery performed 20 years ago (Fig. 1). Despite her previous reduction, she has a very large breast volume and footprint that will be difficult to replicate on the reconstructed left side. She desires nipple preservation and wants to keep her native right breast. We proceed with a left nipple-sparing mastectomy through her previous vertical limb and immediate prepectoral reconstruction using a full-height variable-projection tissue expander (width = 16 cm, height = 16.5 cm, projection = 6.8 cm, and volume = 850 ml) with anterior coverage using an acellular dermal matrix. Twelve weeks later, we exchange her tissue expander for the largest anatomical implant available, a full-height extra-projection 775 ml implant (width = 15.5 cm, height = 16 cm, and projection = 7.1 cm). She also undergoes a contralateral reduction of 300 g to achieve better symmetry. The final result is shown in Figure . Despite using the largest and tallest implant available and reducing the right breast by an additional 300 g, the entire upper pole of the left breast was depleted with significant size asymmetry between the breasts. We discussed multiple sessions of lipofilling to fill this defect but felt that an autologous flap would be more definitive. The LICAP flap was chosen to reconstruct the upper pole of the left breast. An intraoperative photograph is shown in Figure , where an extended flap is dissected based off the known perforators that arise anterior to the latissimus muscle at the level of the inframammary fold as previously described. This flap is rotated on its pivot point and used to reconstruct the upper pole of the breast by suturing it to the underlying pectoralis muscle. The final result 6 months after surgery is shown in Figure , where we have reconstructed the upper pole of her breast with the LICAP flap and have acceptable symmetry between the 2 sides. The patient is discharged the same day, and drains are removed on postoperative day 4. |
A 59-year-old man presented to the urgent care clinic at the School of Dentistry complaining of an upper lip mass for one-year duration. The mass started out as a small bump and had grown steadily since then. 3 months prior to his presentation, a draining parulis developed on the mass. The patient had no history of systemic symptoms such as fever, chills, weight loss, or fatigue. He was aware of a dark-colored “dead tooth” for several decades in the area of concern but denied any previous history of swelling in the area. The patient had recently moved to the United States from Nigeria and had previously been without access to adequate dental care. The patient said that a doctor in Nigeria told him that the lesion was likely cancerous.\nOn exam, there was a large, painless, fibrous, exophytic mass in the anterior maxillary labial vestibule (Figures and ). The base of the mass approximated the apex of tooth #8. A yellow purulent material was observed draining from the parulis (). Tooth #8 was discolored and was confirmed to be nonvital on pulp testing. There was a significant gap between teeth #7 and 8. Tooth #8 was displaced medially and was extruded relative to the adjacent dentition.\nA periapical radiograph revealed a large unilocular radiolucency associated with the apex of tooth #8 (). Cone-beam computed tomography again demonstrated a large cystic-appearing defect in the anterior maxilla with perforation of the buccal and palatal cortices . The lesion extended to the nasal floor on the ipsilateral side.\nThe patient was referred to the oral surgery department for excisional biopsy. After tooth #8 was removed, an incision was made around the base of the stalk that connected the mass to the labial and alveolar mucosa. Sharp dissection was used to free the mass, and the specimen was sent for histopathologic analysis. The mass communicated with a cystic lesion of the maxilla. The cyst was enucleated with a curette and also sent for pathology. Perforation of the cyst through the buccal and palatal cortices was noted during the procedure. Slight undermining of the wound margins allowed for closure with resorbable sutures.\nAt the patient's one-week follow-up (), he was doing very well. He reported minimal pain, no neurosensory disturbances, and no systemic or local symptoms of infection. He and his family were very relieved to learn that the lesion was benign. He was happy with his appearance after having the mass removed. |
A 53-year-old Caucasian woman was referred to our institution because of the suspicion of peritoneal carcinomatosis, raised by the findings of ascites at a transvaginal ultrasound performed as a yearly routine exam; a pre-surgical staging exam with computed tomography (CT) scan show thickening of the gastric walls, multiple omental nodules and ascites ().\nHer previous personal history was unremarkable and she denied any clinical symptom or cancer history.\nAt our hospital, she underwent an esophagogastroduodenoscopy and colonoscopy, with results negative for gastric/colon cancer.\nHer comprehensive metabolic profile revealed mild liver dysfunction with an alanine transaminase of 77 U/L and aspartate transaminase of 71 U/L. When tumour markers were assessed, CA125 demonstrated increased levels of 290 U/mL (normal values <35 U/mL), whereas carcinoembryonic antigen, CA 19.9 and other immunohistochemical markers were within the normal ranges. Serological assessment of HIV, hepatitis C virus and hepatitis B virus were negative.\nTen days later, the patient underwent an ultrasound-guided biopsy () with a diagnosis of suspicious carcinoma from the an unknown primary site.\nAfter 2 weeks, the patient received a CT scan of the thorax (to complete pre-operative staging), demonstrating a spontaneous (with no therapy) dimensional and numerical reduction of peritoneal lesions in the upper abdomen, partially included in the chest CT scan, as well as resolution of peri-hepatic and peri-splenic ascites ().\nSince there was no evidence of primary cancer at pre-operative examinations and the second CT scan revealed a partial resolution of peritoneal implants and ascites without therapy, the suspicion of an infectious disease was raised.\nThe pathological evaluation of the biopsies performed on the omentum and peritoneum revealed the presence of lymphoid aggregates with a central core of epithelioid cells with large eosinophilic cytoplasm, without atypia or mitosis () and with no immunohistochemical marker of oncologic malignancy. Due to the presence of necrotic nodules with histiocytes and giant cells, a Ziehl–Neelsen stain was performed to identify bacilli (), whose presence was then confirmed with molecular assays.\nBecause of the uncertain result of the biopsy and the conflicting results of the 2 CT scans, in order to rule out malignancy with certainty, the patient underwent a laparoscopic surgery in 2 weeks.\nDefinitive histological diagnosis excluded the presence of malignant cells and reported a necrotising inflammation caused by non-tuberculous mycobacteria. Hence, the patient was sent to a hospital with expertise in infectious diseases.\nA follow-up CT scan performed 1 year later confirmed a complete recovery of peritoneal findings. |
An 11-month-old boy was referred to a tertiary center for failure to thrive, poor muscle tone, short neck, kyphosis, and unusual spacing between teeth. He was diagnosed with infantile HPP after repeated low ALP activity test results and radiographic assessment of severe rickets-like skeletal changes and tongue-like lucencies projecting into the metaphyses. He was hospitalized multiple times for pneumonia likely related to musculoskeletal manifestation of HPP, which required treatment with intravenous antibiotics. At age 9 years, he developed persistent headaches; a magnetic resonance imaging (MRI) cranial scan confirmed craniosynostosis with Chiari malformation and cerebellar tonsillar herniation. He underwent craniovertebral decompression, with removal of the posterior arch of C1; a ventriculoperitoneal shunt was inserted to relieve intracranial pressure. He subsequently underwent 2 shunt revisions.\nThe patient experienced multiple fractures, starting at age 17 years, when he sustained bilateral femoral fractures when jumping off a wall; this required bilateral intramedullary rod insertion. At age 18 years, he sustained a right tibial fracture while jumping. Bone healing was delayed, but the fracture eventually healed satisfactorily. At age 20 years, he sustained bilateral femoral fractures when he rolled off his bed during a seizure and required rehabilitation for approximately 8 months.\nOver the first 18 years of life, the patient was hospitalized 8 times for a total of 43 days (Table ). Of these hospitalizations, 5 separate admissions required a stay of ≥5 days; the stay for insertion of a ventriculoperitoneal shunt was 14 days.\nOutpatient specialist visits represented a significant proportion of the healthcare resource utilization by this patient (Fig. ). Most of the outpatient specialist visits required consultation with providers in 12 specialties, including pediatric dentist (38 visits), pediatric endocrinologist (32 visits), neurosurgeon (27 visits), and general pediatrician (19 visits; Table ). Outpatient management consisted of diagnostic imaging procedures (Table ). The most frequent procedures were radiography of the limbs and spine, performed on 18 and 12 occasions, respectively; the patient also underwent 14 MRI cranial scans. Dental surgery and tooth extraction were performed as day case procedures for management of dental carries and malocclusion on 3 occasions. |
A 74-year-old male patient underwent left total knee arthroplasty (TKA) at our institution for degenerative knee osteoarthrosis. He had an uneventful postoperative course with no history of delayed wound healing or persistent drainage. Four years later, he presented with a 2-month history of a gradually developing painless swelling over the anterior aspect of the operated knee; the swelling was associated with a small sinus that was extruding a straw-coloured fluid. He had no history of fever, decreased appetite, or weight loss. He had no other musculoskeletal, respiratory, or systemic symptoms of note. He had no history of antecedent trauma, recent travel, or contact with infectious diseases. The patient is a known hypertensive, but the blood pressure was well controlled with treatment, and he is otherwise healthy. He is a retired teacher with no history of involvement in activities requiring excessive kneeling. He is ambulatory in his community and can walk comfortably with the assistance of a cane.\nThe patient's general physical examination results were within normal limits; positive physical findings were limited to the involved knee. There was an anterior knee swelling involving mainly the prepatellar area, approximately 7 cm in diameter, fluctuant, and not tender to palpation, with minimal surrounding erythema; the erythema was present mainly at the punctum. The punctum was draining a yellowish discharge on pressure (). There was no bony tenderness at the patella, distal femur, or proximal tibia. There was no detectable knee effusion, instability, or crepitus. The range of motion was well preserved (5–110°), as it was a prosthetic knee. It was only painful at the end of flexion as this movement compressed the prepatellar bursa.\nPlain radiographs of the knee showed a prepatellar soft tissue swelling (). There were no obvious bony changes, osteolysis, or loosening at the bone-prosthesis interface. Needle aspiration of the prepatellar bursa yielded 50 mL of slightly turbid straw-coloured yellowish fluid that was sent as per our protocol for cell count determination, microcrystal analysis, Gram staining, and Ziehl–Neelsen (ZN) staining for acid-fast bacilli (). Cultures for aerobic and anaerobic bacteria, Mycobacterium tuberculosis, Brucella, and fungi were requested.\nConsidering the patient's age, chronic presentation, and the presence of sinus, he was admitted with a diagnosis of infected prepatellar bursitis. The primary aims of admission were wound dressing, awaiting aspiration results, and further work-up. Aspiration results revealed normal cell counts, no crystals, and no organisms on Gram stain. To our surprise, ZN stain revealed acid-fast bacilli consistent with typical tuberculous infection; the bacterium was confirmed on culture 6 weeks later as Mycobacterium tuberculosis-sensitive to rifampicin, isoniazid, ethambutol, and streptomycin. Cultures for bacteria, Brucella, and fungi were negative. Aspiration was repeated and yielded similar results. The blood work-up showed normal total and differential white blood cell count and slight elevation of both erythrocyte sedimentation rate (ESR) (80 mm/hr; normal: 30–70 mm/hr) and C-reactive protein (CRP) (8 mg/L; normal: 0–4 mg/L). Tuberculin skin test revealed a negative result (<5 mm induration at 72 hrs), and further work-up including chest radiograph and echocardiogram revealed no evidence of systemic disease. A triple three-phase bone scan displayed normal uptake both at the bone-prosthesis interface and at the patella. An infectious disease consult was obtained. The patient was started on rifampicin (600 mg/day), isoniazid (300 mg/day), pyrazinamide (1500 mg/day), and ethambutol (800 mg/day) for 2 months and continued on the same dosage of rifampicin and isoniazid to complete a 6-month course.\nThree weeks later, the swelling significantly subsided in size and the sinus healed; therefore, the patient was discharged. He was reviewed at 6-week intervals at both the orthopaedic and infectious disease clinics for clinical progression and for any side effects of the medication. At the most recent 6-year follow-up, he was doing well with no evidence of local recurrence or prosthetic loosening (). |
Prior to visiting our clinic, a 19-year-old female with a history of DS presented to an outside ophthalmology clinic for the evaluation of reduced visual acuity. Based on the observations by her family members, the patient had a 1-year history of a marked decrease in ‘visual activities’. The patient's past medical history was significant for DS and sensorineural hearing loss. It was also notable for duodenal atresia repair 10 years prior to presentation. Her family history was significant for only 1 male cousin with LHON. One unaffected member of the extended family had previously undergone documented genetic testing for the LHON mutation, which revealed that a sister of the patient's grandmother was a carrier of the 11778 mutation. Additionally, several maternal relatives of the patient reported positivity for the mutation, although evidence from previous genetic analysis could not be found. Given that all maternal offspring would be expected to be carriers of the disease, there were 25 unaffected carriers of the mutation, of which 9 were male heirs (fig. ) [].\nThe patient was referred to our clinic approximately 1 year after her diagnosis. On examination, visual acuity was 20/200 bilaterally. There was no relative afferent pupillary defect, though Amsler grid testing revealed bilateral central scotomata. Color plate testing revealed 5/6 color plates bilaterally. The patient had 10 diopters of esotropia on straight gaze with full ductions, and the intraocular pressures were 17 and 19 mm/Hg, respectively. An examination of the anterior segment, her lens and the vitreous were unremarkable. Dilated fundus examination demonstrated pale optic nerves and bilateral cup-to-disc ratios of 0.8 (fig. ). The retina was otherwise unremarkable. A generalized neurological examination demonstrated a mild cognitive impairment.\nSix months prior to her visit, the patient had undergone an MRI of the head and orbits for evaluation of compressive, ischemic or demyelinating disease. MRI had revealed an increased T2 signal within the optic chiasm and the optic nerves bilaterally, suggestive of a metabolic or mitochondrial disorder. Subsequent mitochondrial genetic testing was performed via pyrosequencing, and the LHON G11778A mutation was confirmed. In our clinic, the patient was unable to complete a Humphrey visual field examination or tangent field testing; therefore, a Goldmann visual field was administered in order to confirm the presence of central field defects (fig. ). Though the patient had poor fixation, bilateral central scotomata were present. Subsequent optical coherence tomography of the optic nerve head demonstrated a mild thinning of the retinal nerve fiber layer, with an average retinal nerve fiber layer thickness measuring 81 μm OD and 77 μm OS. The average cup-to-disc ratio was noted to be 0.81 OD and 0.75 OS. |
A 4-month-old female Chinese infant was suspected of having convulsive seizures due to an excessive startle reflex on the first day of life and admitted to a local hospital. She was the first child of a non-consanguineous family with no family history of neurological disease. She was delivered vaginally at term with a birth weight of 3450 g and no remarkable antenatal abnormalities or birth history. The Apgar scores for the infant were 10, 10, and 10 at 1, 5, and 10 minutes after birth, respectively. After a course of antibiotics, gastric lavage, and other supportive treatments, she continued to be hypertonic and was discharged home. On the 11th day of her life, the symptoms occurred again at a local hospital. Her convulsive seizures were characterized by cyanosis of the lips, salivation, and hypertonicity of the limbs and lasted about 2 minutes before spontaneous resolution. Because no improvement occurred after treatment with phenobarbital for about 4 months, she was sent to our hospital, where we found hypertonicity in response to sudden auditory stimulus. The patient's clinical diagnosis was subsequently revised to hyperekplexia.\nThe findings on cranial magnetic resonance imaging, full blood count evaluation, C-reactive protein measurement, as well as blood gas and electrolyte analyses were normal. Pulmonary computed tomography suggested left inferior lobe pneumonia. Electroencephalography showed abnormalities with an α-like rhythm in the midline and bilateral frontal areas during both sleep and wakefulness. Several paroxysmal abnormal movements were detected with enhancement of the aforementioned α-like rhythm. In Xiamen Childrenʼs Hospital, physical examination showed a positive result on the nose-tapping test (head-retraction reflex). To exclude epilepsy, video electroencephalography was performed. During the test, some stimuli were presented, but no abnormal waves were induced except for some myoelectric artefacts.\nFor genetic analysis, peripheral blood samples were collected from the patient and her parents. Genomic DNA was extracted for Tiro Whole Exome Sequencing. The possible effects of the mutations on GLRA1 protein function were analyzed using SIFT, PROVEAN, MutationTaster, Mendelian Clinically Applicable Pathogenicity, REVEL, GERP, and phastCons20way. Preliminary genetic tests revealed complex heterozygosity of the GlRA1 gene. One mutation was at c.1030C > T in exon 8 p.R344X,106 (p.Arg344Stop.106), and another was a novel mutation at c.292G > A in exon 4 p.D98N(pAs98Asn) (Fig. ). The c.1030C > T mutation was also detected in her mother's sample, and the c.292G > A was found in her father's sample (Fig. ). Therefore, her diagnosis was finally confirmed as hereditary hyperekplexia with GlRA1 hybrid gene mutations based on the sequencing results.\nThe patient was treated with clonazepam (0.044 mg/kg/d) three times a day after diagnosis with hyperekplexia. Her muscle hypertonia recovered rapidly, and the excessive startle reflex to unexpected stimuli disappeared by the second day of administration of the medicine. The dose of clonazepam was continually applied until the follow-up 2 months later; at that time, her muscle hypertonia was nearly normal and the startle responses were almost resolved. |
A 36-year-old healthy male patient reported to the oral and maxillofacial surgery outpatient clinic at the College of Dentistry and Dental University Hospital, King Saud University, in October 2012. The patient sought medical attention for a slow-growing, painless swelling in the right preauricular region. History revealed that the patient noticed the swelling almost 4 years ago, and since then, it had gradually increased in size with no obvious symptoms or changes to the overlying skin. Upon interviewing, the patient reported no relevant medical or surgical history and mentioned fear of surgery and absence of discomfort as reasons for delaying medical consultation, in spite of an unesthetic facial appearance.\nClinical examination revealed a firm, nontender, nodular, and mobile swelling with apparently normal overlying skin. The swelling extended superoinferiorly from the level of the external ear to the lower border of the mandible and anteroposteriorly from the angle of the mouth to the posterior border of the mandible. There was no lymph node involvement or facial nerve deficit (). Preoperative computed tomography (CT), magnetic resonance imaging (MRI), and fine-needle aspiration cytology (FNAC) were ordered. CT with contrast revealed a well-defined mass lesion in the superficial lobe of the right parotid gland, without any underlying bony erosion and normal-appearing pharynx, larynx, and parapharyngeal spaces. While confirming the CT findings, head and neck MRI further demonstrated a well-demarcated, heterogeneous, mass lesion measuring 10 × 7 × 8 cm in maximum dimension (). FNAC showed numerous scattered groups and clusters of plasmacytoid myoepithelial cells, associated with a chondromyxoid matrix. A provisional diagnosis of PA with no malignant tendency was arrived at based on CT, MRI, and FNAC findings.\nSurgical removal of the right parotid SGT under general anesthesia was planned and explained to the patient. Following informed consent, the lesion was excised completely through ECD, with preservation of all branches of the facial nerve. The right parotid gland was approached using a cervically extended preauricular skin incision. A clearly discernible plane of dissection around the tumor was used for dissecting the tumor mass, without any iatrogenic damage to the facial nerve branches. Owing to the long-standing nature, multiple small feeder vessels had to be ligated circumferentially around the tumor to achieve hemostasis. The intraoperative period was unremarkable, and the patient did not require any blood transfusions (). The resected mass was bilobed and ovoid in shape with a final dimension of 7 × 13 × 7 cm and weighing 1.2 kg.\nHistopathological examination of the excised specimen gave a gross appearance of a partially encapsulated mass containing myoepithelial and ductal proliferation. There was marked stromal hyalinization, squamous metaplasia, and keratinization. Some epithelial islands exhibited papillary configuration, along with large cysts and inflammation. Chondromyxoid changes and fibrosis were evident throughout the tumor. Certain foci of tumor islands were seen approaching and breaking through it. Hematoxylin and eosin (H&E) stained sections revealed a partially encapsulated tumor with variable histopathological features and focal effacement of the fibrous capsule. The tumor typically showed epithelial/myoepithelial proliferation punctuated by chondromyxoid areas. Aggregates of plasmacytoid myoepithelial cells as well as ducal differentiation surrounded by clear myoepithelial cells were evident. Based on the above findings, a final diagnosis of benign PA was reached. While the tumor sections showed no evidence of malignant change, there was extensive squamous metaplasia and keratin cyst formation, which were atypical for PA (). The patient was therefore advised close follow-up, once every month for the first year postoperatively and subsequently once in six months.\nAt 6 weeks postsurgery, the patient had unremarkable wound healing without any neurological deficit of the facial nerve (). As of December 2019, the patient had a recurrence-free follow-up period of 7 years and presented with normal activity of muscles of facial expression, indicating the absence of any long-term facial nerve weakness (). |
A 37 years-old unmarried premenopausal female presented in the emergency of our hospital with complaints of bleeding from ulcerative lesion in the left breast for a couple of hours. The patient had history of ulcer in the left breast for past 2–3 weeks which developed over the underlying huge breast lump. The lump was present for past 5 months but rapidly grew over the last 3 months to current size. There was no history of coagulopathy, trauma, chronic liver disease or history of similar illness in first degree relatives. The patient attained menarche at the age of 15 years. She is a non-smoker and denies history of contraceptive use or alcohol intake.\nOn examination in our emergency there was severe pallor with tachycardia. However the blood pressure was maintained. There was an oozing ulceration in upper outer quadrant of the left breast with underlying huge lump measuring 15 × 15 cm occupying the upper outer, lower outer and upper inner quadrants with deformed shape of the breast (a). The skin surrounding the ulcer was erythematous and edematous. Axilla did not show lymphadenopathy.\nBefore the development of breast lump, the patient had itchy lesion at the same site. The patient unfortunately attributed all these events to that itchy lesion and sought help late. There was no history of headache, shortness of breath, abdominal distension or bone pain.\nOn investigating the patient, she was severely anemic with hemoglobin of 5 gm% with normal total and differential counts. Coagulation parameters and liver function tests were normal. Chest X-ray showed no evidence of metastasis other than dense breast shadow on left side due to huge breast lump(a).\nMammogram revealed homogeneous radiodense lobulated mass occupying whole of left breast with subcutaneous thickening on the background of dense breast (b). Ultrasound (USG) of abdomen and pelvis did not reveal any abnormal findings. Trucut biopsy from the breast mass showed necrotic areas with fibrocollagenous tissue with proliferation of oval to elongated spindle cells showing mild degree of atypia.\nAfter discussion about the possibility of metaplastic versus malignant phyllodes tumor, the patient underwent modified radical mastectomy (MRM). Histopathological examination showed maximum tumor size of 14 cm with increased stromal cellularity, loss of stromal-epithelial balance and frequent mitoses more than 45 per 10 high power fields () typical of malignant phyllodes. All the resected margins, nipple and areola were free of tumor. Lymphovascular and perineural invasions were not identified. Seventeen axillary nodes retrieved were free of tumor.\nPostoperatively patient gained weight and was doing well. Three months after surgery, the patient started developing progressively increasing headache, nausea and vomiting for which she got admitted in our center. There was no local recurrence. However on further investigations, she had a huge cystic lesion with mural nodule in her brain (b,c,d). Contrast enhanced computed tomography (CECT) of chest, abdomen and pelvis showed lesions in lower lobe of left lung (a) of size 3.9 × 3.6 cm of +40 Hounsfield unit (HU) in posterobasal segment and right adrenal gland () of 5.8 × 5.1 cm of +30HU with significant enhancement in post-contrast images. Functional evaluation for the adrenal mass with urinary metanephrines and serum cortisol was negative. Patient refused biopsy from right adrenal gland and lung, but while preparing for burrhole biopsy from brain, patient succumbed to the disease. The patient underwent serial ultrasound of abdomen and pelvis during hospital stay which showed increase in size of 4 cm of adrenal mass in 15 days (tumor velocity). |
An 80-year-old man with known cervical canal stenosis due to ossification of the cervical posterior longitudinal ligament was transferred to an emergency room soon after a falling accident. The patient was alert and had complete tetraplegia, sensory deficits below the C4 cervical sensory level, and progressive restrictive respiratory failure that required assisted ventilation, and, eventually, a tracheostomy. Cervical spine magnetic resonance imaging showed cervical cord compression that was worse at the C3–C4 intervertebral level, and an intramedullary high signal at C3 and C4 vertebral levels, and the patient was managed conservatively. During the conventional weaning process of trials of intermittent spontaneous breathing in the ICU, an unexpected cardiac arrest made both the patient and medical staff reluctant to proceed with further weaning. Fifty-eight days after cervical cord injury, the patient was transferred to our hospital for further rehabilitation. He was fully conscious and received pressure-support mechanical ventilation with back-up rates of 16 breaths. He had total tetraplegia below shoulder girdle muscles with a preserved sensation from C2 to C3 on both sides, and a urinary obstruction. The limbs and trunk were stiff with contracture, and muscle stretch reflexes of the limbs were abolished. Neurological level of injury was C3 according to the ISNCSCI, with a total sensory score of four. There was no sensory sacral sparing, while the deep anal pressure was preserved, and the American Spinal Injury Association scale graded B. Chest roentgenogram and computerized tomography showed bilateral, widespread atelectasis. The patient occasionally showed signs of pulmonary infection. Intensive pulmonary care and rehabilitation, including physical support for expectoration, mechanically assisted removal of tracheal secretions (CoughAssist E70®, Philips Japan, Tokyo), ventilator muscle training, and management of the spasticity of the abdominal wall, resulted in the improvement of the roentgenological findings. His expiratory tidal volume (VT) measured 150 mL.\nAlthough the neurological status according to the ISNCSCI was unchanged, the patient was willing to be weaned from mechanical ventilation as his general condition improved. After informed consent was given, we attempted automated weaning from mechanical ventilation using IntelliVent®-ASV on and after 131 days of injury. IntelliVent®-ASV (Hamilton Medical AG, Switzerland) is a closed-loop ventilation mode that adjusts the pressure support in terms of the percentage of the ideal minute volume (%MV). The ideal MV (100%MV) is calculated from the patient’s height and gender. Based on Otis’ equation [], adaptive support ventilation will select the best VT–respiratory rate (RR) coupling for the optimal work of breathing. IntelliVent®-ASV has interrelated functions: an auto-adjustment for carbon dioxide (CO2) elimination, an auto-adjustment for oxygenation, an auto-weaning tool named Quick Wean (QW), and a spontaneous breathing trial (SBT). The setting of the target %MV is automated based on either the monitored end-tidal CO2 or monitored spontaneous breathing rate. The auto adjustment of fraction of inspiratory oxygen or positive end-expiratory pressure is based on the monitored oxygen saturation fraction of hemoglobin with a pulse oximeter (SpO2) []. The QW mode is an optional automated weaning, and progressively reduces the pressure support, monitors for readiness-to-wean criteria, and provides the option to automatically conduct a fully controlled SBT. In the QW mode, when the spontaneous rate is less than the upper limit of the predicted target range, %MV is automatically decreased gradually to the selected level. To facilitate the recovery of VT by means of the loading work on respiratory muscles, we set the level of support to 70%MV for the QW mode. When spontaneous breathing satisfied the pre-determined conditions of oxygenation and ventilation for 1 min, the SBT mode was used. We operated the SBT mode during the day from 9:00 a.m. to 6:00 p.m.\nIn the QW mode, the duration of 70%MV and SBT time (25%MV) increased gradually (Figs. and ). Twenty-two days after the introduction of the QW and SBT modes in association with intensive pulmonary rehabilitation, SBT was running almost fully during the daytime. At this time, the expiratory VT was increased to 350 mL from 150 mL upon admission to our hospital. Thereafter, we withdrew the pressure support ventilation during the nighttime 39 days after the introduction of the auto-weaning mode, and the patient’s weaning process was completed. |
We discuss a 59-year-old female who presented for planned PICC insertion for chemotherapy administration. Her medical history consisted of sigmoid adenocarcinoma with a Hartmann’s procedure the month before and subsequently commencing chemotherapy a week before. She was a non-smoker and had no familial history of coagulation disorders.\nThe PICC line was inserted into the right arm under ultrasound guidance, without any immediate concerns noted during the procedure. An X-ray revealed the PICC tip to be in a satisfactory position, but potentially a little too deep (). There were no symptoms of chest discomfort or palpitations.\nApproximately an hour later, she developed symptoms of pain and weakness to her left arm (the opposite side). Her arm was clinically assessed as cool to touch with a delayed capillary refill with pulses absent. A computed tomography angiogram (CTA) of her left arm was arranged which revealed an abrupt stop in the left axillary artery ( and ) with reconstitution of the vessels in the antecubital fossa. She was taken to theatre for a brachial embolectomy to re-establish blood flow to her acutely ischaemic arm.\nBrachial access was obtained over the antecubital fossa, the brachial artery was controlled and incised. Proximal embolectomy with balloon catheter retrieved a long segment of thrombus, with resulting good inflow and back bleeding. Post-operatively she was commenced on therapeutic low molecular weight heparin (LMWH).\nShe was subsequently worked up for a cause for this limb threatening pathology. Echocardiogram revealed unremarkable cardiac valves without evidence of an embolic source. A bubble study was also performed which showed no evidence of right to left shunt or patent foramen ovale. Histology confirmed recent blood clot without organization. At follow up she had a good radial pulse and her LMWH was switched to rivaroxaban.\nPICC lines are generally considered safe, with delayed complications including occlusion, accidental withdrawal, infection and thrombotic complications []. Immediate complications are well documented, including those of a cardiac nature due to the tip in contact with the right atrium; atrial or ventricular contractions leading to atrial fibrillation (AF), supraventricular tachycardia or cardiac arrest [].\nIt has been reported that PICC line insertion can affect the right atrium and result in AF, and cease when the PICC tip is retracted [, ]. X-ray has been suggested as the gold standard for post procedural check, but this only gives an anatomical estimate of positioning rather than functional—it has therefore been suggested that electrocardiogram (ECG)-assisted placement is performed to assess for arrhythmias such as AF []. Stroke is the most widely documented risk of AF with the left atrial appendage also being affected and therefore allowing for embolic propagation from the left heart into the arterial circulation []. It should however be noted that other arterial embolization, although only accounting for <10–12% of events, can be just as devastating [, ].\nOur case suggests a subclinical episode of AF from PICC line insertion which resulted development of thrombus. This propagated to the left arm causing an acutely ischaemic limb. The rapid succession of events leading to this is of most interest. Of course there are pre-existing risk factors in our patient that predisposes her to being hypercoagulable (recent surgery and cancer diagnosis). It is likely that these factors all contributed to her overall hypercoagulable status, in addition to the likely arrhythmia acting as a trigger. Unfortunately no post procedural ECG was obtained to confirm AF and our suggestion is of course speculative. No other cause was found on investigatory work-up.\nRight arm venous access resulting in left arm arterial embolism can only be explained by an event affecting both cardiac atria. The right arm sustained no complications and working PICC line would confirm correct entry along the venous system. The possibility of right to left shunt was excluded on investigation as this would cause embolism propagation from the left atrium to the arterial system. Without a physical defect electrical conduction across the cardiac septum is a possible explanation. It is known that an electric connection exists, starting with the PICC triggering arrhythmia in the right atrium and electrical conduction propagating from there to the left atrium via the coronary sinus musculature connection []. Although arrhythmias can communicate between atria, this eliminates the need for a thrombus to develop in the right atria and travel through the lung filter. It is more likely that the thrombus origin is in the left atrium. |
In 2006, a 22-year-old man presented with worsening headache for 1-month duration with phonophobia, photophobia, and worsening lower extremity weakness. A computed tomography (CT) and magnetic resonance imaging (MRI) of the head revealed a large supracerebellar, infratentorial large mass measuring 3.5 × 2.5 × 2.6 cm (). He underwent a suboccipital craniotomy with resection of the mass and a ventriculoperitoneal shunt (VP) was placed due to hydrocephalus (). Pathology from a biopsy taken from surgery confirmed a pineoblastoma.\nThe patient received concurrent chemotherapy and radiation with weekly vincristine and conventional proton radiation therapy for 5 weeks. Afterwards, he was referred for maintenance chemotherapy under pediatric protocol Children’s Oncology Group 99701 regimen B but was ineligible due to his age and previous treatment with vincristine. After a prolonged hospital course which was complicated by pneumonia requiring a tracheostomy and dysphagia requiring a percutaneous endoscopic gastrostomy tube placement, he was discharged to an inpatient rehabilitation facility. He recovered well and his only residual effect from his treatment was a cerebellar gait.\nHe was admitted 9 years later in 2015 at the age of 31 after a seizure episode. He was found to have a left frontal mass measuring 7.2 × 5.2 × 3.4 cm with regional sulcal effacement, midline shift, effacement of the lateral ventricle, and rightward subfalcine herniation. The mass was resected and confirmed a grade 2 atypical meningioma. Post-operatively, he did not have major complications, but he developed intermittent seizures and traumatic brain injury symptoms and was treated with levetiracetam.\nTen years after the initial diagnosis of pineoblastoma and 1 year after the diagnosis of atypical meningioma in 2016, he was admitted for a 5-day history of severe hypogastric abdominal pain and worsening numbness and weakness of his lower extremities. CT and MRI imaging of his abdomen were obtained which showed a large pelvic mass with osteoblastic involvement of the bilateral proximal femurs, pelvis, and cervical, thoracic, and lumbar vertebral bodies ( and ()). Interventional radiology guided pelvic mass biopsies were consistent with his prior diagnosis of pineoblastoma, and no surgical options were available due to the extent of disease. Genetic testing was not performed, as it was not indicated. To treat delayed intraosseous metastasis of pineoblastoma to the pelvis, he was started on the Pediatric Head Start II regimen with five cycles of induction chemotherapy with cisplatin, cyclophosphamide, etoposide, and vincristine with plans for an autologous stem cell transplant in the future. Immunotherapy was not considered, as patient had poor social support and would not have been able to adhere to close clinical monitoring. His chemotherapy course was complicated by pancytopenia, pneumonia, and deep venous thrombosis, and after his fifth cycle of induction chemotherapy, the patient was lost to follow-up. |
A 77-year-old female patient with a past medical history of chronic obstructive pulmonary disease, hypertension, hyperlipidemia who presented to our emergency room (ER) due to worsening of respiratory distress. Four days prior to admission, the patient was hospitalized for management of community acquired pneumonia and upper gastrointestinal (GI) bleed and was later discharged to a rehab center. During a physical therapy session at the rehab facility, the patient developed severe shortness of breath and was found to be hypoxic with an oxygen saturation (SpO2) of 75%, she was placed on a nasal cannula. Presenting vitals in the ER showed a blood pressure of 152/83 mmHg, pulse 137 bpm, respiratory rate of 18/min and SpO2 97% on a 15 litre nonrebreather, and the patient was found to be COVID-19 positive using the RT-PCR test. Initial computed tomography (CT) of the chest pulmonary embolism protocol found multiple small bilateral pulmonary emboli as well as elevated right ventricular to left ventricular ratio of 1.4 indicating right heart strain (). The patient was noted to have expressive aphasia and left sided flaccid paralysis, and an initial CT scan of the head without intravenous (IV) contrast showed no evidence of acute intracranial abnormality. A magnetic resonance imaging (MRI) of the head with and without gadavist intravenous (IV) contrast () done later on Day 1 found a large acute right middle cerebral artery (MCA) territory infarct. The patient was started empirically on IV dexamethasone, antibiotic therapy, aspirin, atorvastatin, and therapeutic enoxaparin. On Day 2, a CT angiography of the head and neck with IV contrast showed an abrupt vessel cut off of the right internal carotid artery at the level of the supraclinoid right ICA, and nonvisualization of the distal right internal carotid artery as well as the right M1 due to a non-occlusive thrombus ().\nThe patient was transferred to an acute care facility, and a transthoracic echocardiogram () showed moderately dilated right ventricle with moderately reduced systolic function, right atrial thrombus in transit and hypermobile atrial septal aneurysm. The thrombus was managed with thrombectomy using the Inari FlowTriever device () and was started on continuous IV infusion of unfractioned heparin. On day 3, a venous doppler of the lower extremities showed a thrombus in the right femoral vein of the distal thigh, gastrocnemius veins, posterior tibial, and peroneal veins. At this point, COVID-19 related hypoxic respiratory symptoms were mild and required only nasal cannula for supplemental oxygen. Patient remained hemodynamically stable until day 10 her condition was complicated by an upper GI bleeding in the form of melanotic bowel movement, she subsequently underwent esophagogastroduodenoscopy (EGD) and found a clot in the anterior bulb, which was treated with epinephrine, clot removal and cautery of a non-bleeding vessel. She was then treated with pantoprazole IV infusion and transfused two units of packed red blood cells (PRBC), while anticoagulation therapy was held. An interdisciplinary decision was made to restart anticoagulation on day 13. On Day 18, the patient had a hemorrhagic conversion of right middle cerebral artery ischemic infarct which was detected on CT of the head (), and heparin infusion was discontinued. Neurosurgery evaluated the patient and she was deemed not a candidate for any surgical intervention. Given her grave prognosis, the patient was transitioned to hospice care on Day 20. |
Our patient is a 58-year-old male with past medical history of hypertension and distant right femur fracture who was involved in a motocross accident suffering left- (1st-12th) and right-sided (7th, 9th, and 12th) rib fractures with bilateral hemopneumothoraces requiring bilateral chest tube placements. The patient also suffered a right intertrochanteric and a peri-implant femur fracture as well as a nondisplaced ulnar styloid process fracture. The patient was transferred from an outside hospital for further care. Upon arrival the patient was remarkably asymptomatic, in normal sinus rhythm, hemodynamically and respiratory stable, maintaining an O2 saturation between 95% and 100% with minimal supplemental O2 via nasal cannula. The patient denied significant chest pain or shortness of breath despite his significant injury burden. ECG findings showed some ST wave abnormalities suggestive of early repolarization. An initial troponin level of 0.15 normalized within 24h of admission. Given the patient's injury pattern and troponin leak in the absence of known coronary artery disease (CAD), congestive heart failure (CHF) pulmonary embolism (PE), or shock, a formal TTE was obtained. TTE revealed severe tricuspid regurgitation secondary to flail anterior tricuspid valve leaflet with preserved right ventricular geometry and systolic function. The cardiac surgery team recommended outpatient follow-up for elective repair in two months with repeat TTE.\nThe femur fracture was repaired on hospital day two under general anesthesia and the patient recovered well. The chest tubes could be sequentially removed without recurrence of pneumothoraces over the course of the admission. On hospital day three the patient experienced an episode of atrial fibrillation with rapid ventricular response that responded well to a single 5 mg intravenous bolus of metoprolol. After a few hours the patient converted back to normal sinus rhythm, in which he remained until the day of discharge on hospital day seven. The patient continued to deny any palpitations, shortness of breath, or radiating chest pain. By the time of discharge, the patient was able to ambulate, and his pain was well controlled with oral analgesics. Unfortunately, by one year after hospital discharge, the patient had not followed up with regard to his newly diagnosed tricuspid regurgitation. |
In November 2012, a 38-year-old male presented with 2-year progressive vision loss in his right eye. He was referred to our clinic for a choroidal lesion with subretinal fluid in the right eye. He had been generally healthy and denied any significant past medical history including diabetes mellitus and hypertension. Since 2010, the onset of the vision decrease, a flat serous retinal detachment in his right eye was detected by a local ophthalmologist. However, no treatment was given, for the diagnosis was unclear at that moment. After several outpatient follow-ups, the disease progressed gradually. One year later, he visited another ophthalmologist for a second opinion. At that time, a choroidal mass with associated serous retinal detachment was detected, yet no treatment was performed, for the diagnosis remained unclear. At his initial visit to our institute, best corrected visual acuity was 20/200 in the right eye and 20/16 in the left eye. Intraocular pressure of both eyes was within normal limits. Examination of the left eye was unremarkable. Right eye anterior segment was normal. Fundus examination revealed choroidal thickening with diffuse retinal pigment epithelium (RPE) changes and inferior exudative retinal detachment (Figure ). Fluorescein angiography showed nonspecific leakage at the level of the RPE and formation of macular edema, combined with thickening of the choroid and serous retinal detachment (Figure ). Ultrasonography showed low-reflective masses with diffuse thickening of the choroid and involvement of optic nerve and orbit (Figure ). A suspicious diagnosis of posterior scleritis or choroidal hemangioma was made. Retrobulbar twenty-milligram triamcinolone acetonide was then given. However, the ocular findings were unchanged. One week later, he received experimental treatment of intravitreal ranibizumab.\nThe patient was loss to follow up until 6 months after intravitreal ranibizumab injection. Magnetic resonance imaging (MRI) of the orbits confirmed extensive choroidal thickening involving the optic disc with a height of 0.37 cm. Two lobulated soft tissue masses were seen around the optic nerve. Fundoscopy revealed deteriorated retinal detachment (Figure ). The patient and his family refused to undergo aggressive treatment, including biopsy. One year later, patient returned to our clinic with visual acuity of no light perception on the right eye. Since the etiology of the mass was uncertain, enucleation was performed after written informed consent was obtained from the patient. Histopathology revealed the choroid, iris, ciliary body and trabecular meshwork infiltrated with malignant cells (Figure ). Orbit and optic nerve were also involved (Figure ). Intra-retinal exudates were noted and the retina was detached with subretinal exudation and some foamy cells. Fibrous membrane was noted between the retina and a diffusely thickened choroid. This was found to be heavily pigmented lymphocytes with small irregular nuclei. Extension of malignant cells to the retrobulbar tissue was also noted (Figure ). Immunohistochemistry showed positive staining for CD5, CD20, CD23, CD38, CD79a, CD45RO, Bcl-2, Kappa, Lambda, LCA, Vimentin and Ki-67 (5%-10% positive cells), whereas CD10, Bcl-6 and Cyclin D1 were negative (Figure ). The diagnosis of primary uveal EMZL was made based on the histomorphology and immunohistochemistry examinations. The patient underwent a systemic work up including positron emission tomography and bone marrow aspiration smears. The results were negative. |
The 2-year-old male patient presented with chief complaint of gradually increasing circumscribed swelling in the left side of the body of the mandible for last 1 year. The parents did not complain any symptom suggestive of pain in the swelling, but the child used to cry on touching it. There was no other associated complaint. On examination, a firm to hard, circumscribed, mildly tender swelling measuring 4 cm × 2.5 cm was found over the body of the left side of the mandible. Computed tomography scan of the head and neck region showed a soft tissue mass eroding bone over the body of the left side of mandible []. The report was suggestive of neoplastic soft tissue lesion involving underlying bone. The patient's blood count along with other hematological and biochemical parameters was normal. He underwent tru-cut biopsy from the lesion. Histological examination of the specimen showed mononuclear blast-like cells arranged in sheets with thin intervening fibrous septa [Figures and ]. The cells were of intermediate size and composed of round to oval nuclei with high nuclear-to-cytoplasmic ratio and prominent nucleoli and scanty basophilic cytoplasm [Figures and ]. Immunohistochemical stain of the paraffin-embedded tissue sections showed that the blast cells were positive for CD45, CD68 and lysozyme [Figures –] and negative for CD3, CD20, CD99, terminal deoxynucleotidyl transferase (TdT), myeloperoxidase (MPO) and CD138. The histological and immunohistochemical findings confirmed the diagnosis of MS with monocytic differentiation. Then, bone marrow biopsy of the patient was done to detect whether bone marrow involvement was present or not but no abnormality was detected at that point of time. The patient was given cytarabine and anthracycline-based induction therapy followed by consolidation with cytarabine alone. On completion of chemotherapy, swelling completely reduced in size. The patient is now being followed up for any local recurrence or systemic relapse at an interval of every 6 months. |
A 32-year-old male with a history of polysubstance abuse presented to the emergency department (ED) with complaints of bilateral feet numbness. The patient stated that he had consumed alcohol the evening prior to the presentation, and upon awakening the next morning, he had been unable to bear weight on lower extremities. He admitted to decreased sensation bilaterally, which extended from the foot to the ankle, more pronounced on the right side. Furthermore, the patient had a 7/10 dull pain sensation radiating from the lower back to bilateral lower extremities as well as generalized body aches. It was the progressive worsening of his symptoms that had prompted his hospital visit. He denied any other aggravating factors.\nIn the ED, the patient's vital signs were found to be remarkable for a temperature of 97.1 °F, respiratory rate of 19 breaths per minute, heart rate of 124 beats per minute, and blood pressure of 146/27 mmHg. The patient denied any history of diabetes, kidney disease, or back pain. He had smoked half pack of cigarettes a day for "a few years", drank a pint of vodka three to four times a week, and used cocaine as well as marijuana daily for "numerous" years. He reported that he had used cocaine and marijuana a "few" days prior to the presentation. The patient's physical examination was significant for a decreased dull and sharp sensation of bilateral feet with intact sensation above the ankle as well as 1/5 muscle strength of bilateral legs and feet. The muscle strength was 4/5 in bilateral thighs as well as upper extremities.\nAn alcohol level screening was done in the ED, which revealed a level of less than 10 mg/dl. Urine drug screen was positive for cocaine, cannabinoids, and opiates. Lactic acid was elevated at 4.2. Urinalysis revealed a specific gravity greater than 1.030 with large bilirubin, proteinuria, and hematuria. Urine microscopy revealed red blood cells of 4-6 high power field (HPF).\nInitial complete blood count revealed a WBC of 20.9 K/UL with 83.2% absolute neutrophils, and hemoglobin and hematocrit level of 21.7 gm/dl and 64.1%, respectively, with a platelet count of 344 K. The patient’s complete metabolic panel was significant for elevated liver enzymes - aspartate aminotransferase (AST): 3,334 U/L (normal range: 15-41 U/L), alanine aminotransferase (ALT): 850 U/L (normal range: 17-63 U/L), alkaline phosphatase (ALP): 151 U/L (normal range: 38-126 U/L); there was renal insufficiency with a creatinine level of 4.16 mg/dl (normal range: 0.7-1.2 mg/dl), urine nitrogen of 32 mg/dl (normal range: 8-20 mg/dl), and potassium of 7.4 mmol/l (normal range: 3.6-5.1 mmol/L) with high anion gap of 21. An arterial blood gas revealed PH of 7.23, PCO2 of 32 mmHg, HCO3 of 14.9 mmol/L, signifying metabolic acidosis with adequate respiratory compensation. CPK levels were markedly elevated at 100,809 U/L. Initial ECG revealed sinus tachycardia with peaked T wave as seen in Figure .\nThe patient received 2 grams of calcium gluconate infusion for cardiac membrane stabilization. He also received insulin and Kayexalate, which corrected his hyperkalemia. The patient was started on IV fluid (IVF) hydration with strict urine output monitoring. Initial MRI without contrast of spine was performed and was only remarkable for mild spondylosis without spondylolisthesis in the L4-L5 region. Neurology was consulted for a more thorough neurological examination, and the patient was transferred to the intensive care unit (ICU) for close monitoring and management.\nIn the ICU, the patient’s symptoms started to worsen and he reported progression of the numbness up to the knee in both lower limbs. He also complained of worsening pain and weakness in the right lower extremity. During the ICU stay, the right thigh became more swollen compared to the left. Urgent CT scan of the right lower extremity revealed right leg edema without focal tissue or osseous abnormality, as seen in Video .\nDoppler ultrasound showed normal arterial flow and no evidence of deep vein thrombosis. The patient started experiencing new-onset right lower extremity weakness. Dorsal pedis and tibial pedis pulses were present bilaterally. At this juncture, there were concerns for acute compartment syndrome. The patient was placed on neurovascular checks every hour. Intracompartmental pressures were measured frequently, which remained low. Also, surgery was consulted for possible fasciotomy.\nDespite aggressive IVF hydration, the patient’s CPK levels continued to rise. CPK levels peaked at 237,025U/L, even though his hemoconcentration had resolved. Creatinine levels continued to increase, and the patient became anuric and IVF hydration was discontinued. Vascular surgery and nephrology were consulted for emergent Uldall catheter placement and urgent hemodialysis, respectively.\nFollowing repeat sessions of hemodialysis, the patient's renal function improved and CPK levels declined. His lower extremity weakness also started to improve. There was a significant improvement in his left lower extremity numbness. The right leg swelling resolved; however, there was only a mild improvement of motor and sensory loss. The patient continued to undergo physical therapy during his hospitalization. He was subsequently discharged to subacute rehabilitation. The patient was advised to continue with outpatient hemodialysis till full recovery of his renal function was achieved. He was also recommended to follow up with neurology as an outpatient. |
A 14-month-old female child was brought by her parents with a progressively increasing swelling over the lower back and weakness of both lower limbs since birth. There were no complaints of bowel or bladder incontinence. She weighed 15kg and her head circumference was 47 cm. There was no history of any CSF leak from the swelling and no prior history of hospital admission for meningitis. On examination, the girl was paraplegic (0/5 Medical Research Council [MRC]). The lumbosacral mass with a normal overlying skin was fluctuant and had a positive transillumination test and cough impulse. The swelling, which measured 45 cm × 40 cm × 35 cm, had obliterated the intergluteal cleft. Due to the huge mass, she was unable to sleep supine. Magnetic resonance imaging (MRI) revealed the classical imaging appearance of a TMC []. A dilated central canal (trumpet-like flaring) of the terminal cord had herniated into a giant meningocele through a spina bifida defect at L4-S2 level. Two separate sacs (sac within a sac appearance) could be seen distinctly on the MRI. There was no chiari, syrinx, or hydrocephalus on screening MRI of the brain and whole spine.\nSurgery was planned for this patient, keeping in mind that her neurological status was unlikely to improve after surgery but mere cosmetic repair and reduction in the size of the mass will allow for better care. The patient had to be induced and intubated in lateral position. To position the patient prone, about 6000mL of CSF was drained from swelling (the meningocele sac) percutaneously with a wide bore needle with simultaneous fluid replacement to prevent hemodynamic instability. Subsequently, the patient was positioned prone and a vertical incision was made. This opened up the meningocele sac and the inner sac (TMC) was clearly observed []. Myelocystocele was then opened and about 1500mL of clear CSF was drained. Continuation of the myelocystocele into the normal central canal cranially could be easily seen at surgery []. Pial sutures (neurulation) were taken with 8-0 nylon after excising the cyst wall. Primary dural reconstruction followed by multilayered closure of the incision was carried out. She was nursed in lateral position for 48h and received intravenous antibiotics for 24h post-surgery. Patient was discharged on postoperative day 5. On follow-up, sutures were removed on postoperative day 10. At 1 and 6 months follow-up, the patient was doing well, although she was neurologically unchanged. |
A 23-year-old woman presented with a 1-day history of the right hemiparesis before admission to our clinic. She was otherwise neurologically intact with normal mental status, sensation, reflexes, and coordination. No hematological or electrolyte abnormalities, underlying pathologies such as vasculitis, or autoimmune diseases such as systemic lupus erythematosus were identified.\nInitial magnetic resonance imaging (MRI) and computed tomography (CT) of the head demonstrated acute infarction of the left basal ganglia, but no subarachnoid hemorrhage [ and ]. A small, high-density spot on CT and high-intensity spot on T1-weighted MRI was shown beside the infarction [ and ]. Subsequent MR angiography and CT angiography (CTA) demonstrated what appeared to be a 2.3-mm fusiform aneurysm with a left LSA origin [ and ].\nThe cerebral infarction of the left basal ganglia was not aggravated on repeat CT or MRI, and neurological symptoms resolved within 2 weeks. However, DSA revealed filling of 7.8 mm of the fusiform aneurysm near the left M1 segment, with the body of the aneurysm separate from M1 []. The aneurysm was diagnosed as proximal LSA aneurysm. Because the size of the aneurysm was increasing and the risk of rupture was increasing, we decided to perform surgical clipping of the proximal LSA itself to collapse inflow into the aneurysm.\nLeft pterional craniotomy was performed. Intraoperatively, the orifice of the aneurysm was identified not on the proximal LSA but on M1. The aneurysm was brown in color and was diagnosed as a thrombotic aneurysm [ and ]. The orifice of the aneurysm was almost entirely covered with thrombus, and the M1-LSA aneurysm was found to have been mistakenly interpreted as LSA aneurysm on preoperative DSA. Some small LSAs and arteries supplying the surrounding brain were seen to be adherent to the surface of the thrombosed aneurysm. Simple neck clipping was unsuitable, because M1 was collapsed with thrombus after neck clipping, so partial thrombectomy was performed with temporary clips on the parent artery without heparin administration [ and ]. After thrombectomy, neck clipping with a fenestrated clip was performed, preserving the parent artery and LSAs, but not preserving one of the small perforator’s adherents to the aneurysm [ and ].\nPostoperatively, right hemiparesis re-aggravated, and a small new infarction was detected on MRI in the basal ganglia beside the previous infarction []. The aneurysm was not filled, and the left M1 and LSAs were well preserved on DSA [ and ]. She was discharged to a rehabilitation hospital with a modified Rankin Scale (mRS) Grade 2, and then discharged home with mRS Grade 0 at 3 months postoperatively. |
A 68-year-old male was admitted to hospital with a history of dyspnea at rest for 2 d, a cough producing mucus, loss of appetite and diarrhoea. Pneumonia was suspected, and the patient received intravenous antibiotic treatment. During the patient's second night in hospital, he was found groggy and walking near his room by the nurses. They took him back to his room and connected a new antibiotic dose to his venous catheter. A few hours later, he was found unconscious, lying on the floor next to his bed. His peripheral venous catheter was connected to a nasal cannula delivering O2 from the wall, with a flow rate of approximately 2 L/min, and the antibiotic dose was found in the garbage can. As post-mortem lividity was present, no resuscitation attempts were performed by the clinicians and the death was pronounced. The body was immediately brought to the University Center of Legal Medicine (Lausanne, Switzerland).\nBefore any manipulation of the corpse, a native CT scan was carried out at around 10 h post-mortem using an eight-row CT unit (CT LightSpeed 8, GE Healthcare, Milwaukee, WI). All scanning parameters are detailed in . A forensic pathologist immediately viewed the native CT images. To evaluate the distribution of gas because of physiological changes in the body after death, the radiological alteration index (RAI) was used as proposed by Egger et al. []. The RAI was based on the analysis of samples from seven sites (heart cavities, liver parenchyma and vessels, left innominate vein, abdominal aorta, kidney parenchyma, L3 vertebra and the subcutaneous pectoral tissues).\nAll images were interpreted in a consensus reading by one board-certified radiologist and one forensic pathologist who were trained in forensic imaging. A post-mortem radiological report was prepared and described all findings from the native CT scan. The native CT scan revealed the presence of subcutaneous, intramuscular and intravascular gas, a pneumoperitoneum, a bloated heart with the right cavities filled with gas and a left pneumothorax ((a–d)). The maximum RAI score is 100, and RAI scores greater than 50 are usually seen in cases of severe changes []. Therefore, given the short post-mortem interval (<12 h), the elevated RAI obtained in this case (75) strongly suggests an exogenous source of gas in the tissues.\nCT-guided gas samples were immediately taken from multiple sites (carotid artery, jugular vein, right auricle, thoracic aorta, pectoral muscle, thoracic cavity, abdominal cavity, scrotum and gluteal soft tissues, (e,f)) according to the protocol for gas analysis described by Varlet et al. [].\nThe external examination revealed massive subcutaneous emphysema with audible crepitations on the entire surface of the body. Additionally, there was a bruise on the left part of the forehead, a small contusion near the left eyebrow surrounded by a purplish-blue bruise and some bruises of different ages on the inferior part of the thorax and on the legs. On the left arm, a venous catheter was still in place.\nSamples for toxicological and biochemical investigations (blood and urine) were collected in S-Monovette® tubes with sodium fluoride or ethylenediaminetetraacetic acid as a preservative (Sarstedt, Nümbrecht, Germany). Biological samples were collected as soon as possible on arrival of the body at the morgue (vitreous humor) and during autopsy (femoral blood, pericardial fluid and urine). During sampling of femoral blood by incision with a scalpel, multiple gas bubbles were visible in the blood ((a)).\nToxicological analysis of the collected samples revealed acetone at a physiological level in the blood, as well as caffeine and paracetamol in both blood and urine. Post-mortem chemistry analysis of the serum samples collected during the autopsy revealed elevated values of C-reactive protein (175 mg/L), consistent with an inflammatory state and a procalcitonine level compatible with a bacterial infection (0.69 μg/L). The results also revealed signs of cardiac dysfunction with a very high level of N-terminal prohormone of brain natriuretic peptide (>35 000 ng/L) and cardiac necrosis with a very high troponin-T level (>56 000 ng/L).\nThe autopsy was performed by one board-certified forensic pathologist and one forensic pathologist in training. Opening of the thoracic cavity revealed numerous gas bubbles in the fatty tissue covering the heart. The pericardium was opened and filled with water, and the heart floated. An incision was made in the right ventricle with a scalpel, and blood with gas bubbles escaped. This technique was performed mainly for training purposes and not for gas sampling, as the gas from the cardiac cavities had already been sampled during CT imaging. The autopsy revealed the presence of numerous gas bubbles throughout the vascular system, even in the small vessels of the brain ((b)). Additionally, changes to the pulmonary parenchyma were consistent with pneumonia. No other major findings were made.\nAn Agilent 6890N GC (Agilent Technologies, Palo Alto, CA) combined with a headspace gas autosampler and equipped with an Agilent Select Permanent Gases column arrangement was used. This column arrangement is specially designed for gas analysis and contains a molecular sieve 5 Å PLOT capillary column (10 m × 0.32 mm i.d.) and a Porabond Q column (50 m × 0.53 mm i.d.) in parallel, which allows for separation of carbon dioxide (CO2). The column temperature was maintained at 45 °C for 13 min. The injector temperature was 100 °C, and the injection was conducted in splitless mode. Helium was used as the carrier gas at a constant flow rate of 8 mL/min. The gas detection and quantification were performed with a thermal conductivity detector set at 150 °C. The system was calibrated for each gas with standard gases of H2S (Multigas, Domdidier, Switzerland), O2 and N2 (from laboratory air), and CH4 and CO2 (Carbagas, Lausanne, Switzerland). With this system, all the gases could be detected in the same run. The gas compositions for the different intracadaver sampling sites are displayed in .\nSamples of the brain, heart, lungs, liver and kidneys were taken during autopsy and stained following a standard haematoxylin and eosin protocol. The heart samples were taken from both ventricles, and the interventricular septum exhibited epicardial vessels deprived of red cells and small intramyocardial haemorrhages. The lung samples taken from each lobe contained numerous clusters of leucocytes, mainly neutrophils, and, to a lesser extent, some macrophages, predominantly in the left lower and right upper lobes. These findings are consistent with the pneumonia suspected on admission to the hospital. There were no significant findings from the other organ samples. Samples of the heart were prepared and analysed for deposition of the plasma antigen fibronectin and the terminal complement complex C5b-9 to look for early cardiac damage, especially the right ventricular ischemia. None of the samples showed reaction for C5b-9, and only small groups of cells stained for fibronectin. Based on these results, cardiac ischemia was not likely in this case.\nIn light of the different results, the cause of death was attributed to a fatal O2 embolism. This was caused by the infusion of pure O2 from the wall into the vascular system through a venous catheter connected to a nasal cannula. Although from a medico–legal point of view the circumstances of the death (accident, suicide and homicide) remained unclear, police investigations led to the suspicion that the patient had connected the nasal cannula to the venous catheter. The wound on the forehead and the bruises observed during the external examination could be explained by a fall from or next to the hospital bed, as suggested by the police. |
A 53-year-old Korean man presented with gingival swelling and multiple small nodules of the right maxilla that involved the buccal attached gingiva from the canine to the second molar. The enlarged gingival surface was pebbly and white or slightly reddish, without hemorrhage or ulceration (). He also had a small mucosal alteration on the lingual gingiva of the left mandibular first molar, similar to the upper lesion (). These had first been noticed one month earlier, and the dentist referred him to our dental hospital.\nPanoramic radiograph revealed nothing but generalized alveolar bone loss and a periapical radiolucency of the right maxillary lateral incisor (). He had received prosthetic treatments in the right upper molar area 13 months ago and in the left lower molar area 7 months ago. Since the lesions had developed several months ago, both had slowly enlarged. Routine laboratory parameters with immune parameters, such as B cells, T cells, CD4+, CD8+ and IgE, were normal. The patient was generally healthy and was not a smoker. For a clinical differential diagnosis with arteriovenous malformation (AVM), squamous papilloma and verrucous cancer, an excisional biopsy was performed under local anesthesia. There was no severe bleeding, and no alveolar bone destruction. One week after the removal of the periodontal dressing materials of Coe-Pak®(GC Co., Tokyo, Japan), the denuded alveolar bone healed secondarily.\nUnder the approval by Institutional Review Board of Seoul National University Dental Hospital, the removed specimen was fixed in 10% neutral formalin, embedded in paraffin, and sectioned into 4 µm thicknesses. Microsections were stained with hematoxylin and eosin, followed by immunohistochemical staining using antibodies targeting Ki-67 and p53 with an indirect triple sandwich method. Upon microscopic examination, the excised mucosa was proliferating in a papillary pattern with acanthosis. Rete ridges were widened and elongated but were not psoriasiform (). Chronic mild inflammation was also observed. There were a few mitosoid cells among the normal keratinocytes in the stratum spinosum (). No brisk mitotic figures were seen in the stratum basale. Ki-67 was immunoreactive in the suprabasal cell layer as well as the basal cell layer, indicating the hyperplasia of keratinocytes (). P53-positive cells were scattered within the epithelium but were few in number ().\nFor the detection of a HPV subtype infection, 5 - 10 mm3-sized specimen from the central portion of the main lesion were processed on the HPV DNA chip assay (MY-HPV chip kit®, MyGene Co., Seoul, Korea) PCR-based microarray system. This HPV DNA chip contains 20 type-specific probes: 7 low-risk types (6, 11, 34, 40, 42, 43, and 44) and 11 high-risk types (16, 18, 31, 33, 35, 39, 45, 51, 52, 54, and 56) with the additional types of 13 and 32. No HPV DNA was detected on these chip slides (). The final diagnosis of FEH was established based on the histopathological findings. The patient was followed-up for 18 months with no signs of recurrence. |
A 33-year-old married, housewife, belonging to middle socioeconomic status from a rural background, who was premorbidly well-adjusted presented with an insidious onset illness characterized by multiple aches and pains since last 8 years. Detailed evaluation of history revealed that about 8 years back she developed whitish vaginal discharge, which was thick, transparent fluid, nonfoul smelling and intermittent initially, which later increased in frequency. Since her early days, she knew from her friends and relatives that “passage of whitish vaginal discharge is associated with the loss of energy and weakness,” and hence she started remaining distressed. Initially, she developed heaviness of head and over the next few months developed pain in the hands, forearm, elbow, knees, and lower limbs. Also would feel tired for the most time of the day. She discussed about her problem with her friends and relatives, who voiced their concern and told her that these symptoms would persist through her life. This led to increase in her distress and somatic symptoms. Over the years, she observed that symptoms of vaginal discharge would increase few days prior to menses and also noted that any change in the passage of vaginal discharge that is, reduction in discharge would be associated with relief or reduction in aches and pains. Further, she observed that symptoms of vaginal discharge would slightly reduce after intercourse that is, the amount of fluid would reduce for 3-4 days and would be associated with slight improvement in pain symptoms too.\nGradually, the vaginal discharge became more persistent and her distress and somatic symptoms increased. She visited many faith healers, traditional healers, physicians, gynecologists and was suggested various treatments, but did not perceive any improvement. She would frequently self-medicate with analgesics, but did not perceive any improvement.\nOver the years, her symptoms became more severe. Three months prior to presentation to our outpatient, she developed fluctuating sadness of mood, lethargy, anhedonia, poor attention concentration, depressive ideations (ideas of hopelessness, worthlessness), further increase in distress due to vaginal discharge, decreased sleep, and appetite. Psychosocial history revealed that since her marriage she had interpersonal relationship problems with her mother-in-law and sister-in-law.\nShe was evaluated in different treatment settings. In the gynecology services of the hospital, she was evaluated for dysmenorrhea, prolapse of the uterus and lower genital tract infection, but no abnormality was detected. In the medical outpatient services she was evaluated for fibromyalgia, anemia, hypothyroidism, but all the investigations did not reveal any abnormality. She was treated with fluoxetine and amitriptyline from the medical outpatient, but did not perceive any improvement. Following this, she was referred to psychiatric outpatient. After detailed evaluation diagnoses of somatoform pain disorder and moderate depressive episode without somatic symptoms were considered. She was treated with duloxetine up to 60 mg/day along with psychological intervention for 6 weeks. The psychological intervention involved sex education, acknowledging her symptoms, addressing her explanatory models, explaining physiological nature of vaginal discharge, explaining psychological nature of her somatic symptoms, addressing the interpersonal relationship problems with mother-in-law and sister-in-law and limiting her help seeking (i.e. doctor shopping). Over the period of 6 weeks, she gradually improved, and her depressive symptoms resolved completely. Her preoccupation and distress with vaginal discharge also reduced, which led to partial benefit in symptoms of sadness and aches and pains. |
This was a 25-year-old male with known spina bifida who presented with a chronic infection of his left acetabulum. He had been previously managed for several years for a non-healing pressure ulcer of the left greater trochanter, having undergone a partial femoral head resection and prior flap placement with subsequent failure. He presented with large volume drainage from a small ulceration over his left trochanter with CT imaging demonstrating an abscess in the gluteus muscle with osteomyelitis in the abutting femoral head. He underwent a left Girdlestone procedure. Intraoperative findings included heterotopic ossification with necrotic bone in the femoral head. Cultures grew MRSA, Proteus mirabilis, and mixed microorganisms. The surgical wound was treated with a Cleanse NPWTi-d utilizing normal saline. Three days later, he underwent partial delayed primary closure over closed suction drains with the placement of a negative pressure dressing over the incision and ongoing wound, as complete primary closure was not possible due to the dimensions of the resulting wound. He was discharged eight days after the initial procedure on ertapenem. He was not readmitted in the first 30 days after discharge.\nAt his one-month follow-up, it was noted that his left-sided osteomyelitis had not recurred nor progressed. At his two-month visit, the wound continued to be clean and closed, with no sign of breakdown. However, at this time, he developed the worsening of a previously existing stage IV right ischial pressure ulcer, which was treated with operative debridement. He has not had a recurrence of his left hip osteomyelitis and his wound is nearly completely healed. Figure below depicts his chronic trochanteric ulcer, the wound after Girdlestone resection, placement of negative pressure wound therapy over the closed incision, and the resultant healing wound.\nPatient 7: right side\nThis was a 29-year-old male with a history of paraplegia who developed several stage IV ischial and sacral pressure ulcers on his right side, resulting in a dislocation of his femoral head on the right and progression of the infection into the acetabulum and iliacus muscle. His ulcer progressed despite appropriate treatment, and he also developed severe protein malnutrition; he was thus treated with a right Girdlestone procedure. Intraoperative findings were significant for necrotic exposed acetabulum and femoral head. Cultures grew MRSA and Staphylococcus epidermidis. The resulting wound bed, including the acetabulum, was dressed with a Cleanse Choice® (KCI, San Antonio, Texas, USA) NPWTi-d utilizing normal saline. Three days later, he underwent a partial delayed primary closure over closed suction drains with the placement of a negative pressure device over the incision. He was discharged 14 days after the initial procedure on doxycycline and trimethoprim-sulfamethoxazole. He was not readmitted in the first 30 days after discharge.\nAt his three-month follow-up visit, his wound was healing well, with no sign of recurrent osteomyelitis on the right side. However, he did have progressive ulceration of his previously existing left greater trochanter ulcer and was found to have invasive osteomyelitis in the left hip. Figure demonstrates the pre-operative ulcer, resection specimen, and resultant healing wound.\nPatient 7: left side\nDue to the success of the right Girdlestone procedure, the patient underwent a left Girdlestone approximately three months later. Like the right side, he had developed a chronic ulcer over the left greater trochanter with subsequent femoral head osteomyelitis. Intraoperative findings were also similar, with a necrotic femoral head and resultant cultures growing no organisms, though previous cultures grew Pseudomonas. The wound was dressed with a Cleanse Choice NPWTi-d (see Figure below). Three days later, he underwent a partial delayed primary closure over closed suction drains with the placement of a negative pressure device over the incision. He was discharged eight days after the initial procedure on doxycycline and trimethoprim-sulfamethoxazole and was not readmitted in the first 30 days after discharge.\nThe patient was readmitted at 60 days with concern for the protrusion of his left distal femur into the ongoing wound bed and was taken to the operating room for excisional debridement and bone biopsy. The biopsy was negative for invasive osteomyelitis. In addition, at month four, he presented with a stage IV ulceration of his sacrum. Adequate offloading, wound care and nutritional support, and intravenous antibiotics were not able to be achieved in the postoperative care of this patient due to numerous factors. He was discharged in this state five days later on trimethoprim-sulfamethoxazole with the intent to heal by secondary intention and has since re-presented with progressive malnutrition and dry gangrene of the toes of his right leg. He has refused ongoing medical care. He has not required ongoing treatment for the infection in either hip and his surgical wounds continued to decrease in size. |
A twelve year old boy presented to us with a history of a gradually enlarging mass in the left popliteal fossa since eighteen months. He complained of restriction of terminal knee flexion, but he had no pain. On general examination our patient had cutaneous lentiges over the back of the neck and on the skin over the back of left knee. Local examination revealed a palpable mass around 10cms in diameter and about 3 cms raised from the surrounding tissues occupying the entire popliteal fossa. The mass was not warm or tender and was not associated with any skin changes. It was non-pulsatile. The flexion of the left knee was terminally restricted as compared to the right knee.\nThe anteroposterior and lateral radiographs of the knee were normal. Magnetic resonance imaging revealed a well defined lobulated STIR/T2 hyperintense lesion measuring 12.8 × 4.8 × 8.8 cms with multiple thick septae within it. Superioly, the lesion was lying in the soft tissue between semimembrinosus, gracilis and sartorius muscles posteriorly and gastrocnemius muscle anteriorly. Inferiorly it was lying posterior to gastrocnemius muscle. The lesion was lying posterior to neurovascular structures in the popliteal fossa. There was minimal effusion within the joint which was thought to be sympathetic in nature. The mass was interpreted as being either a complicated popliteal cyst or chronic hematoma without ruling out the possibility of soft tissue sarcoma.\nWe decided to perform an open excisional biopsy of the lesion which revealed a reddish-brown cystic-nodular mass. The mass was seen to communicate with the knee joint. The cyst was removed and sent for histopatholoical examination. The communication with the knee joint space was sealed with purse-string sutures. The histology revealed papillary projections lined by hyperplasic synovial cells infiltrated by lymphocytes and few plasma cells with hemosiderin deposits and was consistent with a histology of pigmented villonodular synovitis.\nAs we suspected that the disease had originated in the knee and spread to the popliteal space, the first surgery (posterior only) could not have removed the intra-articular origin of the lesion. So we decided to do arthroscopic synovectomy.\nWe performed an arthroscopic synovectomy 3 weeks later. Brownish-red hypertrophied synovium with papillary projections was found in all compartments of the knee was removed and sent for histological examination. The histology from the intra-articular lesion was also consistent with a diagnosis of pigmented villonodular synovitis. At 6 months follow-up, there is no relapse. |
A 3-year-old girl was referred to our hospital 5 days after the trauma, complaining of right elbow joint restriction and pain. She fell onto her outstretched hand while playing at home from a low chair. Her hand was put into a sling by her grandmother, but she hadn't received any other medical care.\nOn our examination, there was mild swelling over the elbow and tenderness over the radial head. On palpation, the radial head appeared to be anteriorly dislocated. She had pain in her elbow and could not do the active motion. The passive movement of the injured elbow was: 100º of flexion, 20º lack of full extension, 70º of pronation, and 50º of supination.\nRadiographs confirmed an anterior dislocation of the radial head with plastic bowing of the ulna, no evidence of fracture (Fig. ). Ultrasound detected fluid in the joint and soft tissue interposed in the humeroradial joint, so we anticipated that closed reduction would be unsuccessful. The patient was brought to the imaging department the following day. Under anesthesia using a laryngeal mask airway, closed reduction was initially attempted; however, that was unsuccessful as we anticipated. Then, a magnetic resonance imaging (MRI) film was done that showed that a structure of low signal intensity (annular ligament) was stuck in the humeroradial joint (Fig. ).\nThe patient was moved to the operative room the following day (7 days after injury). Closed reduction was attempted a second time, but it also proved unsuccessful verified by intraoperative X-ray fluoroscopy. Open reduction through a posterolateral approach was performed. We found the annular ligament was what stopped the radial head from returning to its anatomical location. The annular ligament wasn't damaged but was very stretched. The radial head was found to be protruding below the annular ligament and was incarcerated between the ligament and anterior capsule, similar to a kind of buttonhole effect (Fig. ). The ligament could not be pulled over the radial head when still intact as that made the joint irreducible. We decided to transect the ligament and repaired it subsequently. By pushing the radial head distally and pulling the interposed ligament anteriorly, it returned to its normal anatomical position immediately. After reduction, the elbow seems to be stable in the proper position during elbow joint movement and forearm rotation, so we finished the operation.\nActive range-of-motion exercises were allowed following 3 weeks of immobilization by a long arm plaster splint. At 3-week, 6-week, 3-month follow-up checkups, the movement range of the operated arm gradually improved, and at 6 months after surgery, it was 135º of flexion, full of extension, 90º of pronation, 90º of supination. This was compared to normal, which was 145º of flexion, full of extension, 90º of pronation, 90º of supination (Fig. ). The result was excellent according to the Mayo elbow performance score (100 scores). |
A 15-year-old male presented to us with 8 month history of progressive difficulty in swallowing and slurring of speech. Her mother had noticed that his speech had become progressively unclear with words merging into each other, without any hoarseness or hyper nasality. He had difficulty in moving the morsel of food in the mouth and pushing it into back of throat but he was able to swallow both solids and liquids without any chocking or nasal regurgitation. The difficulty in swallowing and speech was constant without any fluctuations or diurnal variation. There was no history of tremulousnesss, motor weakness, sensory abnormalities or stiffness or slowing of activities of daily living. There was no history of any drug intake, fever, jaundice, joint pains or rashes. The patient was youngest of four siblings and there was no history of a similar illness in his family.\nOn examination, at the time of presentation, the patient was conscious and cooperative. Systemic examination was normal except mild splenomegaly. Neurological examination revealed normal higher mental functions but Myerson's sign was positive. A Kayser-Fleischer ring (KF ring) was present in both the eyes [] and the cranial nerve examination (other than twelfth nerve) was normal. Tongue was normal in size without any evidence of atrophy or fasciculations but tone was increased on palpation []. All the movements of tongue including protrusion and side to side lateral movements were limited; and tremors were noticed on limited protrusion of tongue. Motor, sensory and cerebellar examination were normal except for activated rigidity in the left upper limb at wrist joint and a reduced blink rate (10/minute). On the basis of history and clinical examination and presence of K-F ring, a diagnosis of isolated tongue involvement as a result of Wilson's disease was entertained.\nHemogram revealed hemoglobin concentration of 12.7 gm%. The white blood cell count was 3600 cells/cu.mm (neutrophil 50%, lymphocytes 39%, eosinophils 4% and monocytes 7%) and platelets count was 88,000/mm3. The serum electrolytes and kidney function tests were normal. Serum bilirubin (total) was 0.6 mg% (direct fraction 0.2 mg) and serum proteins were 6 gm% (with a serum albumin of 3.4 gm%), serum levels of AST, ALT and alkaline phosphatase were 45, 55 and 276 U/L, respectively. His prothrombin and activated partial thromboplastin time were within normal limits. Serum ceruloplasmin was low with a value of 5 mg/dL (normal: 20–40 mg/dL), serum copper level was normal with a value of 82 μg/dL (63.7-140.12 μg/dL) and 24 hour urinary copper was high with a value of 65 μg/day (<40 μg/day). Ultrasound of abdomen revealed a reduced liver span (11 cm) with a nodular surface, splenomegaly and a portal vein diameter of 14 mm, suggestive of cirrhosis and portal hypertension. MRI of brain revealed bilateral symmetrical hyperintensities in putamen, caudate, thalamus, midbrain and pons on T2-weighted images []. On the basis of K-F ring on examination, a low ceruloplasmin level, borderline raised 24-hour urinary copper and findings of chronic liver disease on ultrasound, a diagnosis of Wilson's disease with isolated lingual involvement. He was started on penicillamine with subsequent improvement in his speech and tongue movement []. |
A 14-year-old female dancer presented with increasing right knee pain and functional disability without any trauma. Her pain was retropatellar pain that often radiated medially or laterally from the patella, exacerbated by climbing stairs, running and squatting. She was unable to perform any sports activities. Examination revealed peripatellar tenderness and crepitus, full range of motion and the knee was stable. Radiological examination included both MRI and computed tomography of the knee were performed at the beginning. MRI showed osteochondritis dissecans at the area at the superolateral facet of the patella in the deep subchondral area as shown in . This was appropriately treated conservatively with 6 months of reduced load and protected weight bearing without relief of symptoms. She quit sport activity for 2 years and then another MRI was performed . After 2 years we opted for adipose-derived mesenchymal stromal cells (AD-MSCs) intra-articular therapy according to the procedure described by Tremolada et al. []. The patient’s parents were given written information regarding the use of AD-MSCs therapy, including relative risks of this therapy and also relevant treatment alternatives that could otherwise be explored. Postoperative radiological evaluation was performed at 6 months and 12 months after AD-MSCs procedure as shown in and . Postoperative clinical evaluation was performed using IDKC, Lysholm and Tegner scores (). After surgery, patients began a McConnell program of lateral retinacular stretching, patellar taping and vastus medialis obliquus muscle exercises. The patient was encouraged to swim and cycle to reduce impact loading. Using AD-MSC, she reported a complete relief of pain after 1 year from the procedure. Routine MRI follow-up 2 years later showed evidence of appreciable improvement in cartilage volume and osteochondral architecture at the site of injury. Early MRI T2 mapping at 6 months indicated that the area of cartilage regeneration exhibited high water content suggestive of immature cartilage or fibrocartilage morphology. Later T2 mapping at 18 months after the procedure, indicated progressive maturation of cartilage from deep to superficial layers with more hyaline like cartilage morphology. Limitations of the study are lack of arthroscopic intra-articular visualization and classification of the patella lesion. |
An 86-year-old female with a history of metastatic ovarian cancer presented to the ED with painful bilateral lower extremity edema and a left lateral leg ulceration. Her metastatic ovarian cancer had been diagnosed by malignant pleural effusion five months earlier, and she had completed neoadjuvant chemotherapy with carboplatin and Taxol approximately one week prior to this presentation. She was admitted to the hospital and started on cefazolin for left lower extremity cellulitis on hospital day one.\nOn admission, plain films and ultrasound did not reveal any evidence of osteomyelitis, fracture, DVT, or abscess to the left lower extremity. On exam, she had 3+ pitting edema below the knee bilaterally as well as chronic venous stasis changes. The patient also had a venous ulcer (approximately 2 cm in diameter) on the anterolateral aspect of the distal third of her left lower leg. At the time of admission, this venous ulcer had some serous weeping but no purulent drainage or fluctuance on examination. Her initial Laboratory Risk Indicator for Necrotizing Fasciitis (LRINEC) score was 4, suggesting a low risk for necrotizing fasciitis; however, on hospital day 3, her CRP began to uptrend and she became febrile. At this point, her antibiotics were switched from cefazolin to vancomycin to cover MRSA.\nOn hospital day five, the patient was noted to have a new erythematous area over the anterior left knee, inferior to the patella (). Ultrasound revealed a small fluid collection superficial to the patellar tendon in the infrapatellar region measuring 3.3 × 2.5 × 0.4 cm (). The infrapatellar bursa was aspirated and sent for culture. The patient was started on piperacillin-tazobactam, given the patient's immunocompromised status and subsequent risk for atypical and gram-negative organisms.\nAn MRI was performed on hospital day seven (this was delayed due to the patient's pacemaker) but did not reveal any evidence of osteomyelitis. The patient was clinically improved after starting piperacillin-tazobactam, and vancomycin was discontinued on hospital day seven. On hospital day eight, aspirate cultures returned with Pseudomonas aeruginosa; she was stable for discharge at that time and was sent out with a ten-day course of levofloxacin (culture was pan-sensitive) and close follow-up with infectious disease. |
This is a case of a 59-year-old morbidly obese female with situs inversus totalis who presented for a laparoscopic sleeve gastrectomy. Her BMI was 38 (height 4 ft 11.5 inches, weight 188.2 pounds), and she had a lifelong history of morbid obesity and obesity-related comorbidities, including obstructive sleep apnea requiring a continuous positive airway pressure machine, an elevated hemoglobin A1c (5.8) and a fasting blood glucose increasing her risk of developing diabetes mellitus, and degenerative joint disease which significantly impacted her ability to exercise. The main challenges she identified in losing weight involved eating carbohydrate rich foods, overeating during meals and limited activity due to musculoskeletal pain. The patient had made multiple attempts to lose weight through commercial dieting programs but had been unsuccessful. The patient also completed a six-month medically supervised diet through her primary care provider, which also included working closely with a bariatric registered dietician, following a strict diet of about 1800 calories/day and performing modified exercise, about 120 minutes/week. Despite these intense medical weight loss efforts, she was unable to maintain a healthy weight. The patient was motivated to try bariatric surgery after she witnessed the significant weight loss success her daughters had from this intervention. Her greatest hope from the bariatric surgery was to be healthier and to alleviate her obesity-related comorbidities.\nThe patient’s surgical history included cesarean section and evacuation of an ectopic pregnancy. She was a former smoker, quitting over 25 years ago, and has no other history of substance or alcohol use. Family history is positive for obesity, diabetes, hypertension, coronary artery disease and hypercholesterolemia. She did not take any medications, including supplements, except for Ibuprofen 800 mg 1–3/daily for musculoskeletal pain.\nThe patient underwent a comprehensive evaluation and treatment plan prior to the surgery including: psychiatric evaluation and clearance, nutritional consultation with a registered dietician, education about bariatric surgery and pre/post op expectations, routine preoperative labs, UGI, esophagogastroduodenoscopy (EGD), pulmonary function analysis, and a cardiovascular exam. Situs inversus totalis was confirmed with abdominal X-ray and CT, and echocardiogram. Pre-procedural evaluation with UGI revealed mild gastroesophageal reflux observed to the level of the distal one-third esophagus and small sliding-type hiatal hernia. EGD revealed normal esophagus and duodenum. Stomach biopsy was obtained for antral gastritis, and no helicobacter organism was identified. Abdominal ultrasound confirmed fatty liver disease. Pulmonary function analysis including spirometry, lung volumes and diffusion was normal. A pre-procedural cardiovascular exam was performed due to dextrocardia to exclude other structural cardiac abnormalities. 2-D echocardiography with M-mode demonstrated a left ventricular ejection fraction of 60%, no structural abnormalities in the aortic, pulmonic, tricuspid and mitral valve, or right and left atrium and ventricles. Color Doppler and continuous and pulse wave Doppler demonstrated mild pulmonic regurgitation and mild tricuspid regurgitation.\nThe patient discontinued the Ibuprofen more than one month before her surgery and began a low-calorie liquid diet two weeks prior to surgery. On the day of surgery, preoperative antibiotics, Cefazolin, were given within 60 minutes before the first incision. Prior to induction, the patient received 5000 units of heparin subcutaneously and sequential compression device (SCD) boots were placed for deep vein thrombosis (DVT) prophylaxis. No beta-blockers were administered. The patient was placed in the supine position and general anesthesia was induced. A Foley catheter was placed and the patient was supported with positioning devices including: arms on padded arm boards, gel pad under left axilla, footboard with gel pads, hover mat and a bariatric safety belt over patient’s thighs. The abdomen was then prepped and draped in the standard surgical fashion.\nA Veress needle was used in the right upper quadrant to access the abdomen and insufflation was created to 15 mmHg. Veress was removed and replaced with a 5 mm trocar and the scope was placed. Additional trocars were placed in the following position: right and left 5 mm lateral trocars, right and left 12 and 15 mm supra-umbilical trocars. The placement of all retractors and graspers was adjusted accordingly to the mirror image anatomy of the intraabdominal organs. The primary surgeon was positioned on the left side of the patient and the assisting surgeon on right side of the patient. Situs inversus totalis was confirmed with the majority of the liver oriented to the patient’s right, spleen on the right, greater curvature of the stomach on the right and gallbladder on the left.\nThe operating table was placed in the reverse Trendelenburg position. The patient’s right-sided half of the liver was retracted cephalically using a Nathanson retractor to expose the vicinity of the esophageal hiatus. The peritoneum over the cardia was incised using the Ethicon Harmonic scalpel, and the plane between the cardia and the left crus of the diaphragm was opened to expose the right diaphragmatic crus. No hiatal hernia was present. A point 5 cm proximal to the pylorus along the greater curvature of the stomach was marked corresponding to the incisura angularis just proximal to the crow’s foot of Latarjet’s nerve. The vessels along the greater curvature and all the short gastric vessels were sealed and divided using the Ethicon Harmonic scalpel, freeing the greater curvature and the fundus of the stomach. A 42-French bougie was placed and oriented towards the antrum along the lesser curvature (Figure ). The stomach was stapled and divided alongside the tube in a vertical fashion towards the angle of His (Figures -).\nAn Ethicon Echelon Flex triple staple line power stapler with a total of one black, three green, and one gold staple loads was used. All staple loads were 60 millimeters in length with staple line bio-absorbable reinforcement. Hemostasis at the external staple line was achieved (Figure ). The stomach was removed from the abdomen via the left-sided 15 mm trocar (Figures , ). Intraoperative endoscopy was performed using a 5 mm Olympus Ultrathin gastroscope revealing no areas of stenosis, internal staple line bleeding, nor staple malformation or leak seen. Total operation time was 108 minutes. There were no post-operative complications.\nPost-operative care included pain management and DVT prophylaxis with SCD, heparin and early ambulation four hours post-surgery. A UGI was performed on post-op day 2 with normal findings for a post-bariatric surgery evaluation and clear fluids were started. The patient was discharged on post-op day 3 without any complications. Upon discharge, the patient had a treatment plan that was previously discussed that included instructions for diet and medications, including vitamin supplementation and antacids. At her three-month follow-up, the patient was still without complications and very pleased with her weight loss of 46.2 lbs, weighing 142 lbs (BMI 29). |
An 80-year-old female visited our clinic due to involuntary movement on her oromandibular area and both extremities. She was diagnosed with hypertension and chronic small vessel disease about 10 years before and treated with clopidogrel 75 mg and amlodipine besylate 5 mg daily. Other family members including parents, siblings, 2 sons and 3 daughters were healthy and do not have any history of movement disorder. About 2 years before, she first experienced abnormal movement and discomfort on her oromandibular area. She ignored those symptoms as she had removed dental prosthesis 1 month before. However, those symptoms aggravated progressively. About 1 year later, abnormal involuntary movement that characterized by continuous restlessness was started on both extremities and frequent falling to forward was noted when she was walking. The restless movements worsened with stress and emotion, and were progressive, resulting in speech problems and gait disturbance. Caregivers also complained about memory decline and insomnia. On examination, she showed dysarthria and continuous OMD with generalized choreitic restlessness on her both extremities. Choreitic movement was spread to the trunk and affected sitting or standing posture. When she was ordered to maintain a sustained posture or to close her eyes, she could not maintain even longer than several seconds, which presenting motor impersistence. Cognitive function was also impaired, so it was very difficult to concentrate on or conduct doctor’s request. She scored 21 points on the Korean version of Mini-Mental State Examination. On detailed neuropsychological study (Seoul Neuropsychological Screening Battery), her cognitive function was impaired especially in comprehension, calculation, praxis, language and visuospatial memory and frontal lobe function. On laboratory evaluation including complete blood cell counts, routine chemistry, thyroid function, coagulation profile, tumor markers, peripheral blood smear, serum ceruloplasimin and 24 hour urine cupper were within normal range. Anti-nuclear antibody (ANA) was weak positive, but ANA titer has no clinical significance. Other auto immune studies were also normal. Brain magnetic resonance imaging (MRI) shows no specific findings except diffuse brain atrophy (). Positron emission tomography using [18F]-fluoro-deoxyglucose shows severe hypometabolism in both basal ganglia (). Genetic testing for HD revealed 40 CAG repeats on one huntington allele and 12 repeats on the other. |
A 40-year-old lady presented to the Emergency Department with sudden onset severe bifrontal headache associated with one episode of vomiting. She had no loss of consciousness. Her past medical history was not significant except for occasional bouts of headache, which was relieved by analgesics. Her vital parameters were normal - pulse 78/min and blood pressure of 140/80 mm Hg. She was conscious alert with no motor deficit (World Federation of Neurosurgical Societies grade 1). Emergency noncontrast computerized tomography (CT) scan of the brain was performed, which revealed subarachnoid hemorrhage in interhemispheric fissure with right frontal hematoma and extension of the hematoma in the right lateral ventricle and third ventricle []. In the Intensive Care Unit, she received antiedema measures, antiepileptics, and adequate hydration. She underwent CT angiogram of the brain that revealed single, giant, bilobed, and saccular aneurysm in the interhemispheric fissure at the genu of corpus callosum. The distal ACA was unpaired. An aneurysm was arising from the bifurcation of this azygos A2 segment. It was directed anteriorly with the fundus buried in both cingulate gyrus and medial frontal lobe. The teat was directed anteriorly and to the left [Figure –]. A right frontal craniotomy was performed on the 2nd day of bleed. The brain was tense on opening the dura mater. The right frontal hematoma was evacuated. The interhemispheric fissure was dissected out, and the proximal azygos A2 segment was identified. It was traced distally at its bifurcation to identify the aneurysm and the two A3 segments (pericallosal arteries). The neck of the aneurysm was identified, and the entire aneurysm was dissected out using temporary clip on azygos A2 segment for the duration of approximately 5 min. Two permanent Yasargil clips were applied at the neck of an aneurysm - one a straight and other right angled fenestrated clip to obliterate the neck completely. The flow in the distal A3 segments was confirmed using intraoperative Doppler. Postoperative recovery was uneventful. Follow-up CT angiogram done showed complete obliteration of an aneurysm with patency of the parent azygos ACA and distal pericallosal arteries [Figure and ]. |
A 36-year-old male, a seasoned cyclist with no past medical history, presents to the emergency department with complaints of lightheadedness and diaphoresis after a bicycle fall. Patient was participating in a bicycle race when another rider ahead of him fell causing the patient to swerve to avoid him. Patient states that he fell on his left side and hit a tree with his right leg. Patient was wearing a helmet and did not suffer any chest or head trauma. After the fall, he felt lightheaded and diaphoretic and complained of mid back pain. Patient denied any chest pains or shortness of breath. Patient was subsequently brought to the hospital directly following the accident by ambulance.\nIn the emergency department, patient was noted to be in no acute distress; initial blood pressure was 128/69 mmHg with pulse of 65 beats per minute. He was afebrile, not tachypneic, and well appearing with marked right thigh swelling and tenderness to his medial thigh. Given the dizziness and diaphoresis initially, patient had an ECG performed which showed lateral ST segment elevation () and had a subsequent troponin I that was positive, 0.49ng/mL, with a Creatine Phosphokinase (CPK) of 617 U/L.\nThere was initial concern for a possible cardiac contusion, although the patient had no chest wall trauma and thus was admitted for further evaluation. As an inpatient, an echocardiogram was performed demonstrating normal right and left ventricular function and trace pericardial effusion while the patients troponin continued to trend upwards towards a maximum of 21ng/mL. He was loaded with Aspirin and Clopidogrel as well as initiation of a heparin infusion, Lisinopril, and a Beta Blocker. Coronary angiography was subsequently performed demonstrating a spontaneous coronary artery dissection of left anterior descending coronary artery. No further diagnostic study was performed at that time. Further history revealed that he took multiple caffeine Jello shots and drank a large cup of coffee prior to participation in the race. He denied cocaine, amphetamine, or other performance enhancing drug use ().\nThe patient's CPK and troponin trended downwards on conservative medical management and his back pain resolved; therefore a stent was not placed. The patient was visiting from outside the area; discharge planning included repeat coronary angiography in 6 weeks and instructions that he will not be able to perform competitive cycling again. Should his dissection extend at that period of time or patient become symptomatic, stent placement would be considered. Patient was to continue the Aspirin and Clopidogrel until the repeat angiography was performed. Patient was discharged with plans to follow up with a cardiologist in his home state. Multiple follow-up phone calls made us unable to reach the patient and he was subsequently lost to follow-up. |
A 45-years-old woman was admitted with an unprovoked DVT of the left leg. She presented with one day history of spontaneous pain and swelling of the left leg. The pain was mild to moderate intensity, radiating from the groin to involve the whole left leg. She had no previous history of DVT. She was married for five years but had no children. A right ovarian cyst was discovered during workup for primary infertility. She was scheduled to undergo cystectomy and received a two-week course of norethisterone 5 mg twice daily. She received a shot of intra-articular steroid in her left knee for osteoarthritis three months ago, but was well and active and was not taking any regular medications. There was no family history of DVT.\nShe was an average-built woman with normal vitals. Her left leg was grossly swollen but not erythematous, hot or tender, with intact distal pulses. The circumference of the calf, 15 cm below the tibial tuberosity, was 42 cm on the left compared to 34 cm on the right. The rest of the physical examination was unremarkable, apart from mild tenderness and a vague feeling of a small soft mass in the right lower abdomen.\nHer routine investigations and chest X-ray were normal. The venous Doppler revealed extensive DVT of left common and superficial femoral veins (a misnomer, actually a deep vein). Abdomino-pelvic ultrasound showed a 4.5 × 4.0 cm right ovarian cyst. An abdominal contrast-enhanced computed tomography (CT) scan confirmed a long thrombus involving left femoral and external iliac veins (EIV) extending up to the distal part of the left common iliac vein (CIV), raising suspicion of MTS [ and ]. The right adnexal cyst appeared benign.\nMagnetic resonance imaging (MRI) showed compression of the left CIV by the right common iliac artery (CIA) with a subacute venous thrombus distal to the compression [].\nShe was started on a therapeutic dose of enoxaparin and referred to vascular surgery. However, she was managed conservatively as she was doing well. Six months later, a follow-up Doppler showed partial recanalization of common femoral and near-complete recanalization of left EIV and CIV. Later, laparoscopic removal of the right ovarian cyst confirmed a simple serous cyst. |
A 16-year-old female presented with complaints of passage stool or flatus through the vestibule since birth. She had this complaint since birth, and there was no definite history of vulvar inflammation and fecal leakage from vestibule thereafter. Ultrasonography of abdomen and pelvis showed hugely dilated pelvic bowel loops with inability to visualize rest of pelvic structures. There were no other associated congenital anomalies of the VACTERL type. Bowel preparation was performed with a liquid diet for 1 day, and saline enemas the night before and on the morning of operation and child was taken for examination under anesthesia (EUA). At EUA, four external opening were found in the vestibule, one urethral, two vaginal and one in the lower part of the vestibule communicating with the rectum (1st fistula) [Figure and ]. Per rectal examination revealed hard fecolith confined to anterior rectal wall, and there was no rectal stenosis. Probe was passed in the fistula and incision was made on the vestibular side of opening. Fistula was excised as that of low H-type fistula (perineal canal). However on bimanual examination it was found that a fecolith was impacted outside the lumen of the rectum, between the rectum and duplex vaginas at a higher level. Mobilization of rectum was performed through anterior perineal incision revealed two more fistulous communications, one each in the duplex vaginas with tracts going higher up. A lower midline laparotomy was also performed which revealed didelphys uterus, a type III pouch colon having fistulous communications from pouch to right vagina (2 cm × 1 cm in size, 2rd fistula), terminal end of pouch communicating with left vagina (2 cm × 1 cm in size, 3rd fistula) and distally opening as normal anus []. The fistulas were divided, vaginas were repaired and a complete rectum was mobilized. Excision or pouch colon and pull through of normal colonic segment were performed. Postoperative fasting and intravenous antibiotics were continued for 5 days. The perineum was kept clean and dry by frequent local cleansing. In a follow-up at 1-year child has normal looking perineum, normal vagina with voluntary bowel movements, no soiling and no constipation. |
A 66 year-old woman presented with progressively worsening difficulty in breathing over 3 months. She had a productive cough, orthopnea, and left-sided chest pain. Apart from her reported weight loss, she did not have fever or excessive night sweats. Her past medical history was unremarkable but reported that she used to cook indoors using firewood.\nOn examination, she had labored breathing at rest and lower limb edema with no conjunctival or palmer pallor and no finger clubbing. She had a weak pulse, blood pressure of 135/78 mmHg, and raised jugular venous pressure (JVP). There was a dull percussion note on both lung field with diffuse bronchial breathing. The cardiac apex was displaced with normal heart sounds.\nThe hemoglobin and erythrocyte sedimentation rate were 14.6 g/L and 28 mm/hr respectively. The chest X-ray showed complete consolidation of the left lung (Figure ). She was thought to either have an infectious versus malignant process. Sputum analysis was negative for Mycobacterium tuberculosis. Left pleurocentesis was done draining over a liter of yellowish fluid which was exudative and cytology analysis showed scattered degenerating lymphoid cells consistent with an inflammatory process.\nWithin 2 days after admission, she developed a low blood pressure of 89/54 mmHg and muffled heart sounds together with the raised JVP. She was suspected to have pericardial effusion as she was in cardiac tamponade. Bedside echocardiography was done revealing a 3 cm pericardial effusion. And an electrocardiogram showed a slight low voltage sinus rhythm with poor R-wave progression. An urgent pericardiocentesis was done revealing a hemorrhagic effusion which drained over a liter in the course of her hospital stay. The fluid was taken for cytology analysis, and a CT was ordered.\nA CT of the chest and abdomen showed a large enhancing spiculated mass in the left superior and inferior lingula segments of the lung measuring 8.5 cm × 5.6 cm × 14 cm (Figure ). There were ipsilateral and contralateral mediastinal lymph nodes seen in keeping with lung metastasis. There was a thick 1.7 cm right-sided pericardial effusion noted with bilateral pleural effusion (Figure & Figure ). The liver showed multiple hypodense masses in segments II, III, IVA, and V suggestive of hepatic metastasis (Figure ). The cervical and thoraco-lumbar spine showed multiple osteoblastic bone metastases (Figure ). This was evident for metastatic lung malignancy. The pericardial fluid cytology analysis showed a numerous cluster of atypical epithelioid cells with marked nuclear pleomorphism suggesting malignant cells (Figure & Figure ).\nApart from the left pleural and pericardial fluid drainage, she was also managed with diuretics and antipain. As she had symptomatic relief, she was discharged and referred for treatment of the suspected metastatic lung malignancy, but unfortunately, she passed away because of the advanced disease. |
A 19-year-old male presented in the emergency department with one episode of melena per day, for one week. It was associated with vomiting, shortness of breath and palpitations. His hemoglobin level on initial complete blood count was 5.80 g/dL, signifying severe anemia according to WHO guidelines []. His lab parameters on admission are presented in Table .\nImmediately packed red blood cells (RBCs) were requested from the blood bank. On forward typing his blood group was labeled as O positive and his serum showed strongly positive indirect Coomb’s test with a negative direct Coomb’s. On extended 11 cell panel antibody testing, his serum demonstrated pan-agglutination which matched with monoclonal panel cells having anti-Kell, anti-Lub, and anti-Kpb antibodies. On cross match with four O negative and four O positive packed RBCs, +4 incompatibility was seen with all. Meanwhile a detailed history of the patient revealed two distinct episodes of epistaxis in childhood and a family history of his paternal grandmother having an increased bleeding tendency. In view of his past history of fresh frozen plasma infusions, it was interpreted that the patient may have multiple alloantibodies in blood leading to gross incompatibility. Considering the urgency of the situation, one unit of the least incompatible (O negative) packed RBCs was issued after washing with normal saline thrice, to the emergency department. Transfusion was started under strict monitoring by the emergency department physicians. After slow transfusion of around 10 ml blood, the patient started shivering and his temperature spiked to 101°F with tachycardia and hypotension. The transfusion was stopped immediately and the patient was given intravenous antihistamine and hydrocortisone. Meanwhile, he was transferred to the intensive care unit (ICU) where he received intranasal desmopressin and intravenous factor VIII.\nTransfusion reaction workup revealed a grade 4+ pan agglutination in his serum. During repeat blood grouping, forward typing did not demonstrate any reaction to anti-A and anti-B antisera, like a normal O blood group. However, on reverse typing, his serum showed strong agglutination with group O pooled control cells. His post saline wash incompatibility with O negative red cell concentrate showed minor difference from grade +4 agglutination (pre-wash) to grade +3 clumping (post-wash). A fresh RBCs sample from the patient showed negative direct Coomb’s test, while fresh serum sample remained positive for indirect Coomb’s test. This workup strongly raised the suspicion of Bombay phenotype and his red cells were tested with anti-H lectin, which showed no agglutination. This confirmed his blood group as Bombay phenotype. The reactions observed with Bombay phenotype compared to other blood groups, on forward and reverse typing, are illustrated in Table .\nImmediately, voluntary donor pools were contacted in blood banks throughout the country. Overnight, a donor with Bombay negative blood group was arranged from Karachi. The packed RBCs were airlifted to Islamabad maintaining the cold chain. After crossmatching with recipient’s blood showed no reaction, the donor blood was transfused to the patient. Meanwhile, a distant relative of the patient from a nearby city, with Bombay positive blood group, consented to donate blood at our blood bank. Two days later, another unit of packed RBCs was transfused to the patient. His hemoglobin after two transfusions rose up to 7.40 g/dL. As his melena settled down on supportive therapy, an endoscopy was performed that suggested an underlying hiatal hernia. After surgical consultation, the patient was advised to reduce weight and discharged from the hospital, with a scheduled follow-up visit.\nIn view of the patient’s past medical history and family history, during the follow-up visit, a von Willebrand factor antigen, von Willebrand factor functional activity and factor VIII levels were ordered. His von Willebrand factor antigen level was <2.0%, von Willebrand factor functional activity was <4.0% and factor VIII level was 18.5%, consistent with type 3 von Willebrand disease. The patient and his family were counselled accordingly and referred to the hematology clinic. |
A 22-year-old female reported with the chief complaint of difficulty in swallowing and a swelling on the left side of the palatal region. First, she noticed a small swelling intra-orally on the left posterior palatal region 1 year back which gradually increased in size over a period. Initially, the swelling was asymptomatic but later on as the swelling increased in size, the patient experienced difficulty in deglutition. No history of associated trauma to the region was present. The clinical examination revealed facial symmetry, stable occlusion and no limitation in mouth opening. Intraoral examination revealed a fixed, well-defined bony apophysis present submucously on the left side of soft palate just posterior to the tuberosity []. The mass was approximately 1.5 cm in diameter and was hard and tender on palpation. Color and texture of the overlying mucosa were normal without any ulceration and discharge. Based on these clinical findings, exostosis, osteoma or osteoblastoma was suspected.\nOrthopantomogram (OPG) was done which only revealed an increased radiopacity over the left coronoid region as compared to other side probably due to superimposition. Hence, a computed tomography (CT) scan was advised.\nCT revealed a well-defined hyperdense mass measuring 1.0 cm × 1.5 cm arising from the left lateral pterygoid plate with a continuous medullary cavity extending up to left retromolar trigone region []. The cortex of the lesion was continuous with the lateral pterygoid plate. This continuity of medullary cavity of the lesion and lateral pterygoid plate was highly indicative of osteochondroma. Mandibular region and bilateral temporomandibular joints (TMJs) appeared normal.\nAs the lateral pterygoid plate is an unusual site for osteochondroma, other entities such as osteoma and osteoblastoma were considered as differential diagnosis. An incisional biopsy was performed and based on clinical and histological examination; a diagnosis of osteochondroma of left pterygoid process of the sphenoid was made. She was operated under general anesthesia with nasotracheal intubation. Intraoral incision was placed over the palpable mass, medial and posterior to the left maxillary tuberosity and the mass was exposed. Resection of the bony mass was done and the surgical site was closed primarily. The excised mass was ovoid, yellowish white and hard in consistency. The whole specimen was then sent for histopathological examination which confirmed the incisional biopsy finding. Histopathological examination revealed a core of tissue covered by a cartilaginous cap. The cartilage cells were regularly aligned representing hyaline cartilage. Areas of cancellous bone formation were seen extending from this in a perpendicular direction. The degree of mineralization of bone varied at areas. The marrow space showed adipose tissue associated with stromal tissue []. Thus, a final diagnosis of osteochondroma of the lateral pterygoid plate was arrived at. The patient was kept on regular follow-up and no recurrence or any complication has been noticed for the past 3 years of surgery. |
A 38-year-old female, gravida 3 para 2 at 33 weeks of gestation, was hospitalized for preeclampsia with severe features. A viable neonate had been expeditiously delivered yet the patient’s post-partum blood pressures remained severely elevated ranging from 230/130 mmHg to 280/170 mmHg. She had no antenatal care but reported a history of uncomplicated hypertension during her prior pregnancies and tobacco abuse which was stopped 8 months prior. At the bedside, she complained of mild headaches but denied visual disturbances or upper abdominal pain. She was alert and well oriented with a pulse of 80 bpm. There was no hyperreflexia, clonus, papilledema, peripheral edema or signs of pulmonary edema. Her examination was otherwise unremarkable including the absence of renal bruits. Apart from an elevated random urine protein to creatinine ratio of 0.7, the laboratory investigations were within normal limits including serum creatinine, electrolytes, platelet count, liver function and coagulation studies. There were no laboratory features of hemolysis. She was treated with multiple anti-hypertensives over the next 72 h including oral nifedipine, labetalol and clonidine as well as intravenous infusions of labetalol, nicardipine, hydralazine. Magnesium was used for eclampsia prophylaxis. Of note, a single dose of intravenous enalapril was given with a subsequent 60% increase in serum creatinine that returned to baseline within 24 h of discontinuation. Renal artery dopplers (Fig. ) were performed which revealed greater than 60% stenosis of the proximal left renal artery and at least 60% stenosis of the distal right renal artery. Computerized tomography angiography showed approximately 50% stenosis of the proximal left renal artery without stenosis of the right renal artery (Fig. ). At this juncture, in the setting of recalcitrant severe preeclampsia and the mortality risk of impending eclampsia, an invasive strategy for better evaluation and possible intervention was deemed net beneficial. Renal angiography showed 50% stenosis of the left proximal renal artery for which balloon angioplasty and stenting was performed (Fig. ). The right renal artery demonstrated less than 50% stenosis with an insignificant hemodynamic gradient, thus was not stented. Following revascularization, the patient’s blood pressure improved, ranging from 180/100 mmHg to 160/90 mmHg within 48 h, on dual oral antihypertensive therapy. She was ultimately discharged to titrate further anti-hypertensive therapy as an outpatient. |
A 58-year-old male with a history of diabetes mellitus and hypertension presented at our clinic complaining of gradual painless decreased vision in his left eye over the last year. He underwent cataract surgery for both eyes outside our facility 2 years before the presentation.\nThere was no history of pain, photophobia or floaters. His best corrected visual acuity during the first visit was 20/28 in the right eye and 20/2000 (ability to count fingers at 4 feet) in the left eye. Intra-ocular pressure was 18 mmHg in the right eye and 20 mmHg in the left eye. Subjective refraction did not yield better vision in the left (affected) eye.\nOn examination, both eyes showed a quiet conjunctiva with clear corneas and quiet pseudophakia. Anterior chamber was deep and equal in both eyes. Left eye showed dense turbid fluid behind the IOL, with multiple pitting on the IOL, indicating a previous trial of Nd:YAG capsulotomy (). Posterior capsule could not be visualized by the slit lamp. Right eye showed a clear capsular bag with a good view of the fundus. Fundus exam showed mild non-proliferative diabetic retinopathy and dry macula in the right eye and no view of the fundus in the left eye.\nUltrasound biomicroscopy (UBM) showed dense turbid fluid behind the IOL with varied echogenicity, a severely distended capsular bag, and a thick irregular posterior capsule (). Based on the previous findings, a diagnosis of late onset CBDS was made.\nDue to the poor visibility of the posterior capsule and the overblown capsular bag, surgical intervention using a posterior approach was selected to facilitate surgical capsulotomy without disturbing the position of the IOL and to have better access to aspirate the trapped fluid.\nPars plana vitrectomy and posterior capsulotomy were done. After placing three 23-gauge trocars, a corneal paracentesis was made. After IOL tapping, the entrapped turbid fluid escaped to the anterior chamber (). Anterior vitrectomy and posterior capsulotomy were done using a 23-gauge vitrector ().\nDuring the first post-operative visit, the patient exhibited better vision with a visual acuity of 20/125. The patient reached 20/60 vision after 1 month post-operatively. |
A 32-year-old male with no significant past medical history presented to our facility with a history of worsening pain and swelling in the medial aspect of his proximal left lower extremity over the past several years. Anteroposterior (AP) and “frog leg” lateral radiographs of the left lower extremity revealed a dense lesion consistent with dense matrix, adjacent to the medial aspect of the femur (). An MRI of the left lower extremity revealed that the lesion was of extraskeletal origin, arising from the adductor magnus, without any involvement of the femur (, ). The solid heterogeneous mass was located in the posterior compartment of the proximal-to-mid thigh and measured 9.6 × 7.3 × 13 cm in the transverse, AP and vertical dimensions, respectively. Multiple tiny hypointense foci, which corresponded to the calcific density noted on the prior plain radiographical studies, were noted centrally. The mass was otherwise of intermediate soft-tissue intensity on T\n1 weighted images and moderately hyperintense to muscle on T\n2 weighted images. Following intravenous administration of gadolinium, moderate enhancement of the mass, excluding the central portion, was observed. Superiorly and inferiorly, the margins of the mass were indefinite, and the mass demonstrated peripheral vasculature, which was most prominent at the cephalad and caudad margins. The mass was centred within the adductor magnus muscle fibres that were displaced around the mass. Anterolaterally, the mass was very closely approximated to the posterior cortex of the femur and the fascial margins that separated the quadriceps and the posterior compartments (Supplementary videos). No obvious signal abnormality was noted within the femur to indicate invasion or primary osseous origin. Post-excision pathology confirmed the presence of mesenchymal chondrosarcoma.\nIn the interval, surveillance imaging for metastatic disease with CT scan of the chest, abdomen and pelvis was performed. At 18 months, multiple bilateral non-calcified pulmonary nodules concerning for metastasis were identified (). The most prominent pulmonary nodule was present within the posterior right base, measuring 2.3 cm in the greatest dimension and abutting the pleura (). With regard to the abdominal and pelvic series, a new right adrenal lesion appearing as a hypodense pedunculated mass measuring 1.6 cm in the greatest dimension was noted. There was no evidence of osseous metastases. |
A 62-year-old right-handed man with a past medical history remarkable for coronary artery disease was transferred to our institution after worsening weakness and sensory changes involving his left hand. Family and social history were unremarkable. He initially presented to an outside hospital after developing flu-like symptoms for a week, followed by worsening weakness, numbness, and tingling of the second and fifth digits of the left hand. Neurological symptoms rapidly progressed to affect his entire left upper extremity. Electromyography and nerve conduction studies (EMG/NCVs) showed demyelinating features in multiple left upper extremity nerves. Neurological examination showed normal cognition and cranial nerves function. There was marked left arm flaccid weakness and mild distal right hand muscle weakness. Sensory examination showed decreased light touch and pin-prick on the left upper extremity and evidence of mild distal lower extremities weakness and areflexia. General examination demonstrated marked splenomegaly and generalized lymphadenopathy.\nHe was diagnosed with mononeuritis multiplex, and trials of immune-modulating agents [prednisone, intravenous immunoglobulin G (IVIG), and plasmapheresis (PLEX)] resulted in partial and only transient improvement of sensorimotor symptoms. A bone marrow biopsy identified a large B-cell NHL.\nMagnetic resonance imaging (MRI) of the cervical spine and brachial plexus showed no evidence of tumoral infiltration of the cervical cord or brachial plexus. Cerebrospinal fluid (CSF) examination was performed in two different occasions due to concerns of meningeal infiltration yielding with normal results. The patient was subsequently started on induction chemotherapy including two cycles of cyclophosphamide, doxorubicin, vincristine, prednisone, and rituximab, with partial improvement in strength and sensory symptoms. Two months later he developed worsening weakness in the contralateral hand and mild weakness on the distal left lower extremity. Muscle stretch tendon reflexes were absent. Repeat EMG/NCS showed findings associated with demyelinating polyneuropathy and lumbar radiculopathy with denervation potentials. A sural nerve and muscle biopsy showed a mixed axonal and demyelinating neuropathic process with no specific inflammatory changes in muscle specimen. Over the following month he developed gait difficulties and frequent falls as a consequence of worsening weakness in lower limbs. He also reported hyperesthesia and marked pain on his right arm, associated with progressive muscle weakness. No cranial nerve involvement was observed. He received five PLEX and high-dose steroids with limited improvement. Repeated infusion of IVIG did not improve his motor/sensory deficits. He subsequently completed another cycle of chemotherapy with good laboratory and imaging response regarding his lymphadenopathy and splenomegaly. Follow-up CSF examination was normal. Flow cytometry was indeterminate. Contralateral sural nerve and quadriceps muscle biopsy showed non-specific pathological findings. Repeated CSF analysis showed mild elevation of protein content. He received a cycle of intrathecal methotrexate due to concerns of meningospinal tumor infiltration. The patient was restarted on chemotherapy and completed six courses of CHOP-R (Cyclophosphamide, Hydroxydaunorubicin, Oncovin, Prednisone) with only minor improvement on his neurological deficits. Approximately 7 months following onset of symptoms, he developed binocular horizontal diplopia due to a left third nerve palsy followed by left periorbital discomfort and numbness on the left chin, consistent with “numb” chin syndrome. He continued to worsen and finally develop a flaccid, areflexic quadriplegia, with muscle atrophy without fasciculations. Extensive immunologic, metabolic, and infectious ancillary investigations were normal. Repeated brain [including Fast Imaging Employing Steady-state Acquisition (FIESTA) protocol], spine, and brachial plexus imaging showed no abnormalities. Unfortunately, the patient developed multiple systemic complications requiring intubation, tracheostomy, and prolonged intensive care. He eventually died due to sepsis, neutropenia, and multiple organ failure. At autopsy, lymphoma cells infiltration was demonstrated in multiple cranial nerves including left third, sixth, trigeminal nerve root, and bilateral facial nerves. Widespread, patchy, lymphoma infiltration in peripheral nerves, and distal portions of the spinal nerve roots in upper and lower extremities including bilateral median, ulnar, radial, peroneal, femoral, and sciatic nerve were noted. Lumbosacral and brachial plexus roots along with sympathetic ganglia were also affected. Despite infiltration of cervical sympathetic chain, there was not evidence of symptomatic autonomic dysfunction in our patient during the course of his disease. Assessment was further complicated by associated medical complications. There was absence of lymphomatous infiltration in the brain, spinal cord, or meninges. Infiltrative lymphoma cells affected the dorsal root ganglia and spinal nerves in a patchy, irregular fashion (Figures A,B). |
A 61-year-old woman presented with two supernumerary nipples located along the milk line on each side of the upper abdomen. During a few months before referral, the patient had noticed a firm palpable mass in close relations to the supernumerary nipple on the right side (). She had no other symptoms. Bilateral mammogram and ultrasound revealed normal breast parenchyma. Ultrasound of the supernumerary nipple on the right side confirmed a small mass in relation to this nipple, presenting as a hypoechoic, well-defined area, measuring approximately 10 mm in diameter ().\nUpon clinical examination, the supernumerary nipple on the right side presented with inversion and a palpable firm mass underneath this nipple. Ultrasound-guided needle core biopsy (16G) was performed (). The pathology report described elongated epithelial islands composed of cubic cells, with a small centrally located lumen with focally identifiable cuticles. The epithelial structures were surrounded by a dense fibrous stroma. The microscopic analysis indicated the possibility of SyT and the lesion was categorized as a B3-lesion (a lesion with uncertain malignant potential) ().\nFollowing a Multidisciplinary Team Conference, it was decided to recommend resection with a 5 mm rim of normal tissue, which was subsequently performed ().\nMacroscopically the tumour measured 7 x 5 x 5 mm and was described as a firm and grey-white tumour, in close relations to the supernumerary nipple. Microscopically the tumour measured 13 mm in diameter and was localized in the deep part of dermis and underlying subcutaneous tissue with no relation to the epidermis. The tumour was composed of solid trabecular and glandular imitating formations with focal cysts. The epithelium consisted of cells with slightly irregular nuclei and indistinct nucleoli, surrounded by an eosinophilic cytoplasm. The glandular structures were lined by cuboidal cells. The tumour was in close association with the lactiferous ducts and smooth muscle of the nipple (). Foreign body giant cell reactions, due to ruptured cysts, were identified.\nImmunohistochemical analysis showed positive reaction for CK5, CK14, and P63 () in association with the epithelium presenting as solid cords, whereas the glandular luminal cells showed positive reaction for CK7 (). Thus, the immunohistochemical analysis demonstrated the complex nature of this lesion. The diagnosis was SyT based on the pathology report.\nA re-excision was performed due to insufficient rim of normal tissue in the cranial direction in order to minimize the risk of recurrence. |
A 28-year-old female (60 kg; 165 cm; ASA II) presented for bilateral cosmetic breast augmentation. Ten years prior to this presentation she had been diagnosed with MG with respiratory dysfunction and mild generalized weakness and classified as Osserman stage III. At the time of diagnosis she underwent thymectomy. After the operation tracheal intubation was performed and remained for 7 postoperative days due to myasthenic crisis. During preoperative evaluation her neurological examination was normal and there were no symptoms of MG. She was not receiving medication. Preoperative pulmonary function tests revealed her forced vital capacity (FVC) 3.51 L, with 1 s forced expiratory volume (FEV1) of 2.95 L. The patient's baseline vital signs were a heart rate of 76 bpm and blood pressure of 130/70 mmHg. After discussions with the patient about options for anesthesia a decision was made to proceed with TPVB.\nIn order to provide better consistency in the spread of the local anesthetic and produce a more reliable sensory block, we planned to perform multiple level injections TPVB []. On the day of surgery, standard ASA monitors were applied, intravenous access was established, and crystalloid infusion was started. While the patient was in the sitting position, Th2–Th5 spinous processes were identified and marked by palpating and counting down from vertebra prominens (C7). Under aseptic conditions, local anesthesia with 2% lidocaine was performed to skin and subcutaneous tissues. Linear array probe (Esaote MyLab 5, Genova, Italy) was placed longitudinally in sagittal plane at a point 2-3 cm lateral to the midline. Both transverse processes were visualized as two hyperechoic lines and the parietal pleura was visualized as a bright structure running deep to the adjacent transverse processes. If the pleura was not clearly delineated probe was slightly tilted laterally. The distance from the skin to the paravertebral space was measured (38, 41, and 43 mm at Th3, Th4, and Th5, resp.) and a 22-gauge, echogenic peripheric block needle (Pajunk, Germany) was inserted using in-plane technique in caudad to cephalad direction. The tip of needle was advanced until the superior costotransverse ligament punctured. The proper placement of the needle in the paravertebral space was confirmed with hydrolocation after negative aspiration of blood or air local anesthetic was slowly injected. Spread of local anesthetic was visualized with displacement of the pleura anteriorly. The same procedure was repeated at both bilateral Th2–Th5 levels and total 30 mL of 0.5% levobupivacaine was divided equally in paravertebral spaces. During the block procedure verbal contact was maintained with the patient and there were no signs of local anesthetic toxicity. Also no difficulty of breath or desaturation occurred. Loss of sensation to cold and pinprick of chest wall from Th2 to Th6 dermatomes was verified 20 min following block placement. Intraoperative sedation (Ramsay score of 2-3) was provided with continuous infusion of propofol 30–50 mcg/kg/min (total given was 266 mg). The patient remained hemodynamically stable throughout the 115 min of surgery. At the end of the procedure she was comfortable and pain management consisted of oral acetaminophen 500 mg every 6 h as the postoperative routine. She remained pain-free overnight and did not require any opioids for analgesia. She received tenoxicam 20 mg intravenous for a pain score of 4 (visual analog scale (0 = no pain and 10 = the worst pain)) at the postoperative 15 h. Her lung function capacity was assessed after 24 h and FVC was 3.12 L. She was discharged home on the second postoperative day without any complications. |
A 48-year-old previously healthy woman visited our emergency department due to intractable substernal chest pain. Laboratory tests showed elevated serum cardiac enzymes, which indicated non-ST elevation myocardial infarction (creatine kinase-MB=14.8 ng/mL, Troponin I=3.44 ng/mL). She had no risk factors for coronary artery disease including a history of smoking, diabetes mellitus, hypertension, and dyslipidemia. She did not have a family history of coronary artery disease or gestational morbidity. She had visited another hospital due to chest pain 8 days before admission to our institution. A diagnosis of unstable angina was made and a coronary angiogram was performed, which revealed coronary artery lesions involving the left anterior descending (LAD), diagonal, and ramus intermedius coronary arteries (). Her intractable chest pain persisted despite medical treatment including aspirin and the continuous infusion of heparin and nitroglycerin by our emergency department. A coronary angiogram was performed again and demonstrated aggravated coronary lesions () and multiple luminal narrowings even in the left internal thoracic artery (ITA). Because of the intractable chest pain associated with the rapidly progressing nature of the coronary lesions, the patient was referred for urgent myocardial revascularization. She underwent off-pump coronary artery bypass (OPCAB) using a skeletonized right ITA graft as a blood source. The patient was given an initial dose of heparin (1.5 mg/kg) and periodically received supplemental doses to maintain an activated clotting time longer than 250 seconds during OPCAB. Because her skeletonized right ITA was too short to reach the target left coronary arteries, a segment of the reversed saphenous vein was anastomosed to the end of the right ITA in an I-fashion and grafted to the diagonal and LAD coronary arteries, and the ramus intermedius using a sequential grafting technique. Upon opening the coronary arteries, the lumen was almost completely filled with diffuse fresh thrombi. When the fresh thrombi were removed through the coronary arteriotomies, neither coronary artery dissection nor atherosclerotic plaque was found. Protamine was not given at the end of the procedure. The patient was heparinized in the intensive care unit to maintain an activated clotting time longer than 150 seconds, and anticoagulation therapy using warfarin and aspirin was started at 1 day postoperatively. A angiogram performed 2 days postoperatively demonstrated patent grafts (). Under a suspicion of thrombotic disorder, laboratory markers such as lupus anticoagulant, anticardiolipin antibody, anti-beta2Gp1 antibody, complement 3, complement 4, and fluorescent antinuclear antibody, were evaluated postoperatively. She was diagnosed with APS after a positive result for lupus anticoagulant. She was discharged on the 6th postoperative day on warfarin and aspirin. A 1-year postoperative angiogram was performed and demonstrated completely resolved LAD lesions, a patent graft to the ramus intermedius and diagonal coronary arteries, and a competitive flow pattern in the graft to the LAD coronary artery ().\nA 36-year-old woman with a 6-month history of chest pain visited our hospital due to chest pain of increasing frequency. She had been diagnosed as having systemic lupus erythematosus (SLE) at the age of 20, and had been treated with corticosteroids. Secondary APS manifested as recurrent abortion, an episode of acute pulmonary thromboembolism developed 2 years after the diagnosis of SLE, and anticoagulation therapy was added. Her family history included an acute myocardial infarction in her elder brother at the age of 34 years. Her electrocardiogram and serum level of cardiac enzymes were normal; however, she did have a positive treadmill test. A preoperative coronary angiogram demonstrated a completely occluded proximal LAD coronary artery and multiple stenotic lesions in the obtuse marginal coronary artery (). She underwent OPCAB using skeletonized bilateral ITA grafts. The right ITA was divided at the proximal section and was anastomosed in a Y-fashion to the side of the left ITA. The left ITA was anastomosed to the LAD coronary artery, and the right ITA was anastomosed to the diagonal and obtuse marginal coronary arteries in a sequential fashion. An early angiogram performed on the 1st postoperative day revealed all patent grafts. She was discharged on the 7th postoperative day on warfarin and aspirin. A 1-year postoperative angiogram was performed and demonstrated that all grafts remained patent (). |
A 74-year-old female presented to the outpatient department of neurosurgery with painless, gradually progressive scalp swelling over left frontal region and weight loss for past 1 month. There was no history of trauma or fever. On examination, she was emaciated and had a 6 × 4 cm solitary, tender, soft to firm, fluctuant, non pulsatile swelling in the left frontal region. A 2 × 2 cm solitary lymph node in right posterior cervical region was noted. ESR was 64 mm at first hour. Mantoux was reactive with 18 mm at 72 hours. Chest radiograph was normal. Sputum for AFB was negative. Her immunological status was assessed by serology for HIV, serum albumin levels, fasting blood glucose (FBS), and glycosylated hemoglobin (Hb1 Ac). HIV was nonreactive, serum albumin – 4 g/dl, FBS - 80 mg/dl, and Hb1 Ac – 5%. She had uncontrolled hypertension with a systolic murmur and diastolic dysfunction. CT scan brain showed a bony defect in the left frontal region with soft tissue collection in left subfrontal region with irregular bony destruction []. MRI showed 6 × 4 × 4 cm lobular, heterogenous, multiseptated, mixed signal intensity extradural lesion with thick peripheral enhancement in left frontal bone []. The differential diagnosis included metastasis to the frontal bone, and intraosseous meningioma. Fine needle aspiration cytology (FNAC) of the scalp swelling was performed which on analysis showed epithelioid cell granulomas surrounded by lymphocytes with areas of caseous necrosis suggestive of tuberculosis []. Assuming it as a primarily medical disease, a non-surgical management was planned. The lesion was tapped with 20-gauge needle and around 50 cc of thin, straw-colored fluid was withdrawn; she was started on anti-tubercular treatment for attempted conservative treatment in view of uncontrolled hypertension and cardiac dysfunction. However, the fluid recollected within 24 hours and the plan was changed in favor of surgical debridement under scalp block anesthesia, with a left frontal curvilinear incision over the swelling. The bone margins were nibbled off and the lesion along with sequestrum cleared till the dura mater. The specimen was also sent for cultures (including aerobic, anaerobic, fungal, and TB culture). The Gram's staining showed plenty of polymorphs, with no organisms. The smear for Acid Fast Bacilli was negative both by Ziehl-Neelsen and fluorescent staining using Rhodamine- Auramine stains. Based on the histopathological report, the patient was started on anti-tubercular treatment from second postoperative day, which included isoniazid (5 mg/kg body weight/day), rifampicin (10 mg/kg body weight/day), pyrazinamide (30 mg/kg body weight/day), and ethambutol (15 mg/kg body weight/day). Patient was well for about a week's time when she developed vomiting and her liver function tests showed elevated serum alkaline phosphatase, SGOT, and SGPT. Anti-tubercular drugs were modified and she was started on Tab. Levofloxacin 500 mg, Ethambutol 600 mg, and Inj. streptomycin 750 mg daily. The patient improved with a well-healed scar and no swelling of the scalp. Culture for tuberculosis was positive after 6 weeks of incubation by radiometric BACTEC 460 TB system. After liver function tests returned to normal values, first line anti-tuberculosis drugs were reintroduced which were tolerated by the patient. The isolate was identified as Mycobacterium tuberculosis complex by NAP test (r-nitro-a-acetylamino-b-hydroxypropiophenone) and was sensitive to all first-line anti-tubercular drugs. At the end of one-year follow up, the patient was doing fine with no recurrence of the swelling. Imaging at 1 year following the institution of treatment showed calvarial defect on the left side, with a sunken skin flap. No lesion or abnormal enhancement was noted [Figure –]. |
A 35-year-old man under influence of alcohol fell in a ditch and sustained an injury to his right upper extremity. Clinical examination revealed swelling around the elbow and upper forearm. There was obvious flexion deformity of 30. There was no neurovascular deficit. There was no evidence of compartment syndrome. The patient’s elbow joint was evaluated carefully and showed no coronal instability on physical examination. Plain radiographic studies revealed a fracture middle one-third of radial shaft with a posterior-lateral dislocation of the radial head (). Attempts at reducing the radial head under sedation in the emergency ward were unsuccessful. An above elbow slab was put in the right extremity.\nHe was taken to the operating room the next day. An attempt was made to close reduce the radial head under fluoroscopy, but this could not be achieved. He then underwent open reduction and internal fixation of the radial shaft fracture through a Henry approach. The oblique fracture of radius was reduced and stabilized with a 7-hole 3.5 mm low contact dynamic compression plate on the volar aspect. Fluoroscopy in the operating room showed good reduction of the fracture but persistent widening of the radiocapitellar joint and the radial head dislocated posterior and laterally. Attempts to reduce the radial head with close reduction were unsuccessful. Next, the elbow joint was approached through Boyd’s approach, there was thick fibrocartilaginous soft tissue between radial head and capitellum. This was partially torn off annular ligament lying between radial head and capitellum. Once this soft tissue was removed, the head was reduced. However, it was still dislocating on 60–30 pronation. Hence, a radiocapitellar wire was put through radiocapitellar joint. X-ray showed good reduction of his radial head on the anteroposterior and lateral views (). Postoperatively, the limb was immobilized in a splint at 90 of flexion and 45 of pronation. The radiocapitellar wire was removed at 3 weeks; then, he was started on gentle range of motion exercises after k-wire removal. At 6 months follow-up, range of motion of the elbow was 30 of extension to 120 of flexion and near-normal forearm pronation of 70 and supination of 60 (). On examination, he had no pain over the right forearm and his wounds were healed. At his last visit, 1 year after the operation, he had excellent range of motion with no major complaints. Radiographs showed that the fracture was healed in good alignment and the radiocapitellar joint remained reduced (). The patient reported no pain and was able to resume his normal daily activities, including working as a pharmacist. |
A 32-year-old man was admitted to our hospital with appetite loss. He had a history of traumatic transverse cervical spinal cord injury at the C5 level due to suicide attempt at the age of 18. As a result of cervical spinal cord injury, he was paralyzed in the lower body. Contrast-enhanced computed tomography (CT) revealed a late-onset traumatic diaphragmatic hernia with strangulated ileum (Fig. ). The small intestine, transverse colon, and omentum were displaced into the left thoracic cavity, and some portions of these organs showed a decrease in blood flow. Left lung collapse and a compressed right lung with mediastinal shift were evident. The patient underwent emergency surgery. After replacing the incarcerated organs to their original positions, scattered areas of necrosis were identified in the small intestine, transverse colon, and omentum (Fig. ). By using interrupted sutures with non-absorbable 1–0 monofilament, the diaphragmatic orifice was closed. Wedge resection with primary closure was performed for the colonic necrosis in two places. Partial resection, 45 cm long, with end-to-end anastomosis was performed for the small intestine. The necrotic omentum was removed (Fig. ). In addition, a gastrostomy tube was placed since delayed initiation of oral intake was expected. The patient developed severe septic shock postoperatively. Treatment-resistant critical hypotension with non-compensatory tachycardia developed, likely due to parasympathetic nervous system damage related to the cervical spinal cord injury. On postoperative days (POD) 3 and 6, cardiac arrest occurred. Fortunately, he was rescued by cardiopulmonary resuscitation with administration of large doses of vasopressin and catecholamine. However, peripheral vasoconstriction, increased intra-abdominal pressure, and ischemia of the gastrointestinal tract developed, which resulted in colonic anastomotic leakage with diffuse peritonitis, abdominal wound dehiscence, and collapse of gastrostomy on POD 6 (Fig. ). The patient was unable to undergo surgical repair because of his poor general condition with continuing severe septic and neurogenic shock. Therefore, he underwent AVS through the open abdominal wound and it was the first procedure at the intensive care unit. The procedure of AVS was as follows: 1. the open wound and peritoneal cavity were rinsed with normal saline and necrotic and/or contaminated tissues were debrided (Fig. ); 2. wound dressing materials (DUOACTIVE® ConvaTec, New Jersey, USA) for protecting healthy skin around the open wound were patched along the abdominal wound in piecemeal fashion so as to adjust dressing materials to the complicated shape of the wound (Fig. ); 3. two drainage tubes with multiple side holes, up to 30 cm from the tip, were placed in the abdominal cavity through the open abdomen and the enteric contents were suctioned through the drainage tubes using a Continuous Suction Unit MERA Sacuum (Senko Medical Instrument Manufacturing CO, Tokyo, Japan) set to 50–75 mmHg continuous negative pressure; and 4) the entire wound was filled with saline-moistened gauzes and covered with polyurethane drape (Fig. ). The colonic anastomotic leakage showed gradual healing over the course of 2 months, followed by contraction and closure of wound dehiscence (Fig. ). Because the gastric fistula remained, a gastrostomy balloon catheter was placed through the gastric fistula. The patient resumed oral intake on POD 112 and left the hospital on POD 190 with the gastrostomy balloon catheter and without incisional hernia. |
Francesca (pseudonym) is a 21-year-old young-adult girl with RS. She was born in a non-consanguineous marriage. An uncomplicated pregnancy and a full term normal delivery at a hospital were reported. Regular immunizations were carried out. At birth, her weight and height were normal. Gross motor, fine motor, social and emotional and language milestones were normal during the first 3 years of life. She had normal development up to 20 months when the clinical regression of RS began and she went through the four typical RS stages. The molecular analysis of MECP2 confirmed the presence of the mutation associated with RS (precisely R306C). When she started the intervention, she was 8 years old and she was classified as being in clinical stage IV of RS.\nBy the age of 8 years, she could walk and run, but she did it for hours while turning inside a room. The baseline examination showed that she could not speak any meaningful words, whereas she used to narrate small sentences earlier on. The mother also reported her lack of attachment with family members, her inability to hold, pick up or grasp things in her hands. She would keep both palms of her hands, one over the other, and would move or rub one hand over the other. Teeth grinding was also reported. She was unable to indicate her need for daily activities such as toilet, passing stools, or for food. Marked cognitive and communicative delays were noted. Features suggestive of severe mental retardation were reported. She had no eye contact and characteristic stereotypied hand movements.\nThe intervention started at the age of 8 and Francesca followed 4 sessions each lasting 50 min for each week for a period of 3 years. In this first period, she learned pre-training abilities. At the end of the pre-training (at the age of 11) phase, she started the reading and writing program to begin communicating. She followed 4 sessions lasting 50 min for each week for a period of two and a half years (92 weeks plus holiday time) for the discrimination of words and the bi-univocal correspondence between word and image and between image and word. When she was 13 and a half years old she started the training of separation of words into syllables and reconstruction which continued again for 4 sessions lasting 50 min for each week for a period of 1 year (55 weeks plus holiday time). At the age of 14 and a half, she started the training of separating syllables into letters and reconstruction. This training, again for 4 sessions lasting 50 min for each week, lasted 6 months (24 weeks). When she was 15 years old, she started the construction of sentences training and real communication. She is continuing to communicate even now. |
A 66-year-old female patient came to our service in August 2019, complaining of blurred vision at night for about 11 years. She had experienced binocular cataract surgery successively 2 years ago. Vision examination revealed 20/25 in the right eye (OD) and light perception in the left eye (OS). The fundus photography of her OS revealed characteristic RP fundoscopic findings, which consisted of attenuated retinal arterioles, a waxy pale optic disk, and bone spicule pigmentation. The OD fundus examination indicated similar ghost retinal vessels and retinal pigment clumping; however, the difference was that several spared retinal arterioles which supplied the posterior pole of the retina, and a yellow-white optic disk were observed (shown in Fig. ). Optical coherence tomography (OCT) examination of the OD indicated a relatively normal thickness of each layer in the macular region and the full thickness of macular lutea was 227 μm, yet the atrophic retinal neurosensory layer and retinal pigment epithelium were detected in the peripheral area. In addition, epiretinal membrane existed. Nevertheless, the OCT image of the fellow eye showed atrophy of the entire retina, and the full thickness of macular lutea was only 114 μm (shown in Fig. ). To further identify the spared vessels in the OD, we performed an examination of OCT angiography (OCTA), which revealed that the spared vessels might come from the posterior ciliary artery system but not the central retinal artery. But in the fellow eye, the vascular structure is normal (shown in Fig. ). The visual field measurement exhibited a tubular visual field in the OD eye as well as full blindness in the contralateral eye (shown in Fig. ).\nThe patient was diagnosed with binocular advanced RP based on the examination results above. Considering her stage of RP and available therapies, she was eventually treated with neurotrophic agents, by which we hoped to slow the disease progression. |
A 30-year-old male was admitted to neurosurgery ward with a history of a head injury following road traffic accident. There was no history of loss of consciousness, vomiting, ear bleed, or seizures but the history of bleeding from nose present. On examination, his vital signs stable, he was conscious, well oriented to time, place, and person. His Glasgow Coma Scale (GCS) was 15/15. Pupils were 2 mm bilaterally and reacting to light. No any cranial nerves, motor, and sensory deficit detected. A plain computerized tomography (CT) was done which revealed bilateral frontal contusion with a fracture of bilateral frontal bone with involving the right frontal sinus with mild depression of posterior wall of the sinus with patchy pneumocephalus []. He was put on Antibiotic, anticonvulsant, and analgesics and patient was discharged 3rd of admission. At the time of discharge, the patient was intact without a focal neurological deficit and with no cerebrospinal fluid (CSF) rhinorrhea and GCS was 15/15. A month later, he complained of severe frontal headache with CSF rhinorrhea and progressively deteriorating neurologically then readmitted in neurosurgery ward and on admission his GCS dropped to 12, meningeal signs were found and he became bradypnea and repeat noncontrast CT (NCCT) head revealed tension intraparenchymal pneumatocele on the right frontal lobe with rounded or oval in configuration, measuring on average 3–4 cm in diameter with mass effect and communicating with bilateral lateral ventricles, basal cisterns, and subarachnoid spaces []. Under general anesthesia, a burr-hole was placed in the right frontal region and evacuation of tensed air subdural space was irrigated with normal saline and shifted to the neurosurgery Intensive Care Unit. Next day after the surgical treatment, he improved and became awake. Subdural drain was removed. A repeat NCCT head was done after 48 h which showed resorption of frontal pneumatocele with minimal subarachnoid and intraventricular air []. CSF rhinorrhea stopped.\nThe patient underwent a conservative treatment and observation. On the 5th day after the operation, an episode of hyperthermia up to 38°C developed. On the 10th day, nasal liquorrhea resumed. On the background of the therapy, a sustained clinical improvement was achieved. The patient refused to undergo proposed surgical intervention, aimed at dura defect closure in the posterior wall of the frontal sinus. And discharge on request and gone to any private multispecialty hospital and where watertight dura was closed and cranioplasty was performed. The postoperative period was uneventful, and CSF was without signs of inflammation. In 10 days after surgery, the patient was discharged. At his 6 month follow-up, he has no neurological deficit and no signs of rhinorrhea and meningitis. |
A 69-year-old Chinese woman was presented with left waist pain for 1 month. She had no fever or other pain. There were no abnormal results in the blood routine examination, renal function, and urine routine examination. And the result of urine culture was negative. A preoperative computerized tomography (CT) scan and intravenous pyelogram (IVP) revealed bilateral incomplete duplex kidney and ureter (Fig. ). The stone of calyceal diverticulum was located in the upper kidney of left incomplete duplex kidneys by CT scan (Fig. ). In the meanwhile, IVP and CT scan revealed that a ureterocele was located in the left ureterovesical junction (Fig. ).\nThe patient had hypertension and type 2 diabetes mellitus for 5 years. The risks of surgery and general anesthesia were relatively higher. The diameter of the stone in the calyceal diverticulum was 12 mm by CT scan and it’s difficult for the stone to pass out spontaneously. The patient and her family wished to remove the stone because they were extremely worried about stone-related complications. Moreover, they wouldn’t like to perform second operation and anesthesia due to relatively higher risks. Therefore, we planned to make an attempt to remove the ureterocele and calyceal diverticulum stone in one operation.\nThe contraindications for surgery in such cases mainly included untreated urinary tract infection, severe urinary tract stricture, and anesthetic contraindications like cardiopulmonary dysfunction. Relevant examinations such as lung-function testing and Holter electrocardiogram were conducted. We also paid special attention to the results of urine routine and urine culture. When we made sure all these examination results were normal, we decided to perform the surgery.\nFlexible ureteroscopy with the holmium laser was conducted for solving the ureterocele and stone of calyceal diverticulum. To begin with, the ureterocele was confirmed in view of ureteroscopy and the holmium laser was used for the resection of ureterocele. The ureterocele resection surgery was completed in 10 min and no complications like bleeding occurred intraoperatively. Then, we made an attempt to remove the stone of calyceal diverticulum. During the operation, we found the calyx neck of calyceal diverticulum where the stone was located had obvious stenosis. It took us a lot of effort to find out the stone because of calyx neck stenosis. Fortunately, the stone was discovered when calyx neck stenosis was incised internally by holmium laser. After dilatation of the narrow calyx neck, a 200-μm holmium laser through flexible ureteroscope was used to make the stone fragmented gradually and carefully. The parameter of the holmium laser was set at a power of 0.8 J and a pulse frequency of 10 Hz. Higher power and frequency might cause renal damage and hemorrhage because the calyceal diverticulum stone was near the edge of the kidney and the renal cortex was thin by CT scan. In order to prevent the formation of ureteral steinstrasse postoperatively, a nitinol stone basket was used to remove large stone fragments as soon as possible, and the smaller powdered fragments were flushed out of the diverticulum using an automated irrigation pump. At last, the pelvicalyceal system was examined once again to ensure no large remaining stones. The overall surgical time was controlled in 60 min for the purpose of preventing postoperative infections. A double-J ureteral stent was inserted and remained in place for 1 month. The symptom of left lumbar back pain disappeared post the operation and no complications were developed during the placement of the stent. There were no stone residents observed by CT scan before removing the ureteral stent 1 month later (Fig. ). |
A 29-year-old man presented with a 1-year history of pain in left ankle, with nocturnal exacerbation, which was relieved by the use of acetylsalicylic acid or anti-inflammatory drugs. The clinical examination indicated tenderness of the medial face of the left ankle without swelling. Plain radiographs showed a lytic area, homogeneous, smaller than 1 cm, associated with a perifocal dense bone reaction evoking osteoid osteoma of the distal third of left tibia [].\nThe CT confirmed a hypodensity associated with peripheral sclerosis, located at the distal third of the left tibia, near to the tibio-talar joint [Figures and ].\nThe bone scan showed uptake of radiotracer in the lower third of left leg [], confirming the diagnosis of osteoid osteoma. Due to its deep location, near to the tibio-talar joint, and small size of the lesion with important peripheral sclerosis as shown in the scan [], it was decided to perform an intraoperative isotope detection and to guide the surgical excision. We used a gamma probe counter, which is a scintillation counter used for isotope detection. It is equipped with a collimator connected to the electronics box by a flexible cable of 3.5 m []. The results are displayed digitally in counts per second (cps). The gamma probe counter has the advantage of being in contact with the uptake seat and being oriented to the higher counting rate. This will be displayed later with sound, demonstrating the localization of osteoid osteoma. Three hours before surgery, the patient received intravenously a marked molecule, dihydroxymethylphosphonate (HMDP) which is a bone matrix tracer. In the operating room, we were able to guide the surgical resection through the radiodetection displayed on the electronic box. The patient was taken in decubitus position under spinal anesthesia; the surgical approach was guided by a percutaneous tracking with the probe. After incision and bone access, the osteoid osteoma location was confirmed by probe counting that indicated a major mounting point located in the posterior and interior distal quarter of left tibia. Recording series, inch by inch in the four cardinal directions, allowed mapping the radiolabeling around the optimal point of fixation []. We performed an excision of a bone window of 1 cm / 2 cm around the hyperfixant seat; we recognized the absence of abnormal signal on the residual bone. The postoperative course was uneventful, marked by absence of pain after 24 hours of the surgical procedure. Then, the patient was immobilized by plaster cast for 3 weeks. Pathological examination confirmed the diagnosis of osteoid osteoma with the presence of the nidus. After 24 months, the patient was asymptomatic, and the bone scan performed did not indicate any recurrence of osteoid osteoma. |
A 39-year-old woman, who had been taking medication of quetiapine as an antipsychotics for depression, experienced mild dysarthria and visited the department of neurology in our hospital. Her symptom was diagnosed as drug-induced lip dyskinesia, which disappeared in a week. Screening head magnetic resonance imaging (MRI) at this time revealed stenosis of the left MCA with no brain parenchymal lesions (), why she was consulted to our department. We performed angiography, confirming moderate M1 portion stenosis (Figures and ). SPECT showed no apparent laterality in CBF, thereby we decided to observe her with no treatment.\nSeven months later, the patient experienced mild weakness and numbness in her right hand and visited our department. Although MRI showed no apparent ischemic change in her brain, arterial spin labelling (ASL) of MRI detected the decrease of CBF in the left cerebrum (), which was thought to well correspond for her symptoms. She was admitted and treated with an antiplatelet agent. Two weeks later, she still complained of numbness in her right hand; thereby, we decided to perform left STA-MCA anastomosis to prevent deterioration of her symptoms. Preoperative SPECT showed no apparent laterality in CBF (). On operation, left temporal craniotomy was performed, and the parietal branch of the STA was anastomosed with the M4 portion on the temporal lobe (Figures and ). The intraoperative course was uneventful, and the patient recovered from anesthesia without any new neurological symptoms\nPostoperatively, her speech was normal until postoperative day 1 (POD1). On POD2, she exhibited mild speech disturbance, which worsened day by day finally resulting in complete motor aphasia on POD6. Her comprehension was kept normal. On POD3, generalized convulsion occurred, which ceased quickly by diazepam, and levetiracetam was initiated. On the same day, she presented with mild weakness of right upper extremity, which improved gradually and disappeared on POD7. MRI and CT showed no ischemic or hemorrhagic changes, but ASL and SPECT revealed remarkable increase of CBF in the left cerebrum (Figures and ), by which the symptoms were diagnosed as CHPS. Despite the treatment with strict blood pressure and the administration of edaravone and minocycline, complete motor aphasia remained unchanged on POD21. MRI showed no abnormality except slightly hypointense changes on T2 weighted images and FLAIR (). At this point, the patient was discharged partly because of the request from the patient, and we continued to follow her in outpatient visit. One month after the surgery, the patient started to utter words that were not fluent, when SPECT and MRI showed normalization (). Thereafter, the improvement of her speech was slow, and totally more than three months was taken for full recovery after the surgery. |
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