Complete Resolution Of Symptoms Research Articles

Acalculous cholecystitis in the setting of immunoglobulin a vasculitis: a case report with literature review

Immunoglobulin A vasculitis (IgAV), also known as Henoch-Schönlein Purpura, is the most common childhood vasculitis which presents with a tetrad of symptoms: palpable purpura, abdominal pain, arthralgias, and renal involvement. Gastrointestinal involvement has been reported with findings including abdominal pain, pancreatitis, intussusception, ischemia, and gallbladder involvement. Our patient had the classical presentation of IgAV with additional gallbladder thickening found on imaging. He was managed conservatively for his symptoms and gallbladder thickening. This case adds to the six prior cases that we found in our literature search reported of gallbladder thickening alongside IgAV. However, this was the only case that was managed conservatively resulting in a complete resolution of symptoms.

Electrochemotherapy with bleomycin as an effective local treatment for Kaposi's sarcoma: a case report.

An elderly female patient with a long-standing history of Kaposi's sarcoma of the lower limbs was referred to the Surgical Department after the subsequential failure of multiple lines of systemic chemotherapy. The patient was also complaining of increasing symptoms including intractable pruritus, which negatively impacted her quality of life. She underwent palliative electrochemotherapy with bleomycin (15 g/m 2 ) on the sarcomatous lesions of the left foot and ankle, which lead to complete clinical response and resolution of symptoms; no adverse events were reported. Electrochemotherapy is a valid option in the palliative treatment of Kaposi's sarcoma, as it may lead to satisfactory clinical response and symptom control.

Impact of Objective Colonic and Whole Gut Motility Data as Measured by Wireless Motility Capsule on Outcomes of Antireflux Surgery.

Studies show higher rates of dissatisfaction with antireflux surgery (ARS) outcomes in patients with chronic constipation. This suggests a relationship between colonic dysmotility and suboptimal surgical outcome. However, due to limitations in technology, there is no objective data available examining this relationship. The wireless motility capsule (WMC) is a novel technology consisting of an ingestible capsule equipped with pH, temperature, and pressure sensors, which provide information regarding regional and whole gut transit times, pH and motility. The aim of this study was to assess the impact of objective regional and whole gut motility data on the outcomes of ARS. This was a retrospective review of patients who underwent WMC testing before ARS. Transit times, motility, and pH data obtained from different gastrointestinal tract regions were used in analysis to determine factors that impact surgical outcome. A favorable outcome was defined as complete resolution of the predominant reflux symptom and freedom from antisecretory medications. The final study population consisted of 48 patients (fundoplication [n = 29] and magnetic sphincter augmentation [n = 19]). Of those patients, 87.5% were females and the mean age ± SD was 51.8 ± 14.5 years. At follow-up (mean ± SD, 16.8 ± 13.2 months), 87.5% of all patients achieved favorable outcomes. Patients with unfavorable outcomes had longer mean whole gut transit times (92.0 hours vs 55.7 hours; p = 0.024) and colonic transit times (78.6 hours vs 47.3 hours; p = 0.028), higher mean peak colonic pH (8.8 vs 8.15; p = 0.009), and higher mean antral motility indexes (310 vs 90.1; p = 0.050). This is the first study to demonstrate that objective colonic dysmotility leads to suboptimal outcomes after ARS. WMC testing can assist with preoperative risk assessment and counseling for patients seeking ARS.

Phlebotomy in Secondary Polycythemia: Does It Improve Clinical Symptoms? a Systematic Review

Introduction: Secondary polycythemia (SP) is the increased number of red blood cells in the body that occurs due to elevated erythropoietin levels usually secondary to chronic hypoxia. Treating the underlying cause is the mainstay of treatment of SP. Phlebotomy is rarely used in patients with SP, especially those with symptoms related to hyperviscosity. We present a systematic review assessing the effectiveness of phlebotomy in SP. Methods: Following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines, a comprehensive literature search was conducted on PubMed, Cochrane, and Clinical trials.gov using MeSH terms and keywords for "Polycythemia, Secondary " AND " Phlebotomy " in July 2022. We did not place any time constraints. Our search produced a total of 232 records, and duplicates were removed. After screening and removing irrelevant and review articles, we included a total of 17 original articles and case reports reporting phlebotomy as a treatment for SP in adult patients. The data were collected for baseline characteristics of the participants and efficacy and safety of the intervention. Results: A total of 86 patients from 17 articles were evaluated. (Table 1) The mean age of participants was 51 years and 89% (n=65/73) were males. Most common cause of SP was chronic obstructive pulmonary disease (COPD) 94.4%, followed by congenital cyanotic heart disease (3.4%), high altitude erythrocytosis (1.1%), and obesity hypoventilation syndrome (1.1%). Most common reported symptoms were dyspnea, confusion, headaches, exercise intolerance, and suppression of hypothalamic-pituitary-testicular axis. Two case reports also reported chorea secondary to SP while there was one case report each for hemi-retinal artery occlusion, angina attacks and portal hypertensive gastropathy. Majority of the studies had a goal hematocrit of <55%. There was subjective improvement in clinical symptoms in 85% (n=73) of the patients while 2.3% (n=2) felt worse after phlebotomy. None of the patients (9/9) with suppression of hypothalamic-pituitary-testicular axis had improvement in hormonal profile after phlebotomy. One patient developed hemiparesis, stiffness, loss of dexterity, chorea after phlebotomy with the symptoms resolving when hematocrit went back to baseline >62.5%. One patient died a week after phlebotomy due to respiratory issues. Conclusion: Phlebotomy results in clinical improvement in symptoms in patients with secondary polycythemia. Majority of the patients report either complete resolution of symptoms or at least subjective improvement. Goal hematocrit is usually <55%. Phlebotomy, although rarely used, may be considered a safe and effective treatment option for symptom management in SP, although its utility in objective improvement needs further prospective studies. Figure 1View largeDownload PPTFigure 1View largeDownload PPT Close modal

Single Center Experience with Off-Label Benralizumab in Refractory Hypereosinophilic Syndrome Demonstrates a Satisfactory Safety and Efficacy Profile

Background: Hypereosinophilic syndrome (HES) is characterized by blood and/or tissue eosinophilia resulting in end-organ damage. HES can be primary clonal (in which the eosinophil is part of clonal hematopoiesis), secondary (due to eosinophilic stimulating cytokines) or idiopathic (I-HES). A careful diagnostic work-up for primary causes, as well as vigilant surveillance for end-organ damage should be performed to establish an underlying and treatable diagnosis. Regardless of the cause, treatment of symptomatic HES is mandatory. Treatment may consist of steroids, tyrosine kinase inhibitors, hydroxycarbamide, or interferon, with variable responses. Long-term use of steroids leads to osteoporosis and infections, and treatment with steroid sparing drugs might be hampered by other side-effects. More aggressive treatments such as alemtuzumab is efficacious but severely immunosuppressive. Several antibodies targeting IL-5 or its receptor have been successfully applied for eosinophilic asthma but only mepolizumab is registered by the FDA and EMA for HES. Benralizumab, an anti-IL5R antibody, was efficacious and safe for treatment of HES in a phase-I/II trial. A randomized phase-III trial comparing benralizumab with placebo is currently recruiting. We have treated 15 patients with off-label benralizumab for severe refractory HES or HES with unacceptable steroid dependency. Methods: 15 adults (8 with I-HES, 6 with secondary (lymphocytic variant) HES and one patient with HES secondary to sarcoidosis; age range 19-69 years; 10 male, 5 female) from our institution were treated with benralizumab at a monthly dose of 30 mg after giving consent for off-label use. All had absent HES translocations by FISH, and no evident underlying diagnosis was present after extensive diagnostics including bone marrow examination with cytogenetics and molecular diagnostics. They all were refractory or had contra-indications to conventional treatment, or steroids could not be reduced to an acceptable dose. All had elevated eosinophil counts at diagnosis above 0.6 x 109/L (range 0.32-90 x109/L, median 8.3 x 109/L) except one patient who had a level of 0.32 while on 30 mg prednisolone per day. All patients had at least one organ affected by HES, including 2 patients with myocarditis. Three patients had previously received mepolizumab in a HES dose, 2 of whom were refractory and consequently stopped while one had an allergic reaction necessitating cessation. Median follow-up time after starting benralizumab was 22 months (range, 1-34 months). Results: In terms of efficacy, 13 patients had a reduction of their eosinophil count to below 0.05 x 109/L after one dose, one patient had a reduction from 8.13 to 0.72 x 109/L, with a count that is still decreasing while stopping all other medication. Eosinophil count decreased from 12.38 to 1.45 x 109/L in another patient, but benralizumab was stopped due to the development of MDS, which was most likely the cause of the eosinophilia despite normal cytogenetics and molecular diagnostics at time of diagnosis. All but 2 patients were off steroids completely after 4 months. All but 2 patients experienced a clinically relevant improvement of organ function, mostly to complete resolution of objective symptoms or signs. Interestingly, the 2 patients who did not experience clinically significant reduction of symptoms, despite reduction of peripheral eosinophil counts, both had colitis as one of the cardinal disease symptoms. In one, the diagnosis was revised to inflammatory bowel disease with secondary eosinophilia. In terms of safety, 2 patients experienced mild headache for a few days after the benralizumab dose, and 1 patient had a mild malar rash after the first dose. One patient stopped due to the development of MDS necessitating other treatment and one stopped due to refractoriness. Interestingly, this patient subsequently had an excellent response on mepolizumab, on both clinical symptoms as well as reduction of eosinophil counts. All patients with objective clinical benefit remain on treatment. Conclusions: We report on the largest single center cohort of patients treated with off-label benralizumab for HES. The use of this drug has proven a safe and effective treatment for HES regardless of the underlying cause. It reduces eosinophil counts, reverses organ-damage and use of steroids in most patients. Side effects are infrequent and mild and no severe adverse events were observed.

INNV-23. MANAGING CENTRAL NERVOUS SYSTEM MANIFESTATIONS OF CHRONIC GRAFT VERSUS HOST DISEASE FOLLOWING HEMATOPOIETIC STEM CELL TRANSPLANTATION: A CASE REPORT

Abstract Central nervous system (CNS) manifestation of chronic graft versus host disease (cGVHD) is a rare, difficult to diagnose phenomenon that can follow allogeneic hematopoietic stem cell transplantation (HSCT). CNS-cGVHD typically onsets 2-31 months post-transplant, involves diffuse infiltration of inflammatory cells, demyelination, and cerebral vasculitis, and is associated with an extremely poor prognosis. We present a case of a 37-year-old man who developed seizures, myopathy, bilateral upper and lower extremity weakness, confusion, and irritability almost 2 years after HSCT for acute myeloid leukemia (AML). CNS-cGVHD diagnosis was confidently determined via magnetic resonance imaging (MRI) demonstrating enhanced focal white matter lesions, cerebrospinal fluid (CSF) studies demonstrating elevated globulin (12.7 mg/dL; normal range: 0-6 mg/dL) and elevated immunoglobulin G (IgG) synthesis rate (13.9 mg/d; normal range: ≤ 8 mg/d), and a right brain biopsy demonstrating encephalitis, perivascular inflammation, microglial activation, and gliosis with predominately intramural and perivascular CD3+ T-cells, CD4+ and CD8+ cells. Complete resolution of neurological symptoms, including seizures, was achieved with a combination of steroids, ibrutinib, intravenous immunoglobulin (IVIG), and anti-seizure medications. Improvements to CNS-cGVHD was further indicated by normalized MRI studies without abnormal enhancements and improved CSF studies with reduction in globulin levels (11.3 mg/dL) and complete normalization of IgG synthesis rate (≤ 0 mg/d). It is imperative that CNS-cGVHD be considered in patients that present with neurological signs post-transplant. An early, confident diagnosis generates prompt intervention and as demonstrated, can lead to complete resolution of neurological symptoms.

NIMG-96. SPINAL MENINGEAL MASS LESION: A RARE PRESENTATION OF FOLLICULAR LYMPHOMA

Abstract OBJECTIVE We aim to describe a case of follicular lymphoma that presented as a durally-based thoracic spine lesion manifested as rapidly progressive back pain and meningismus. BACKGROUND Follicular lymphoma is one of the most common forms of non-Hodgkin’s lymphoma; however, it is rare for follicular lymphoma to present as a spinal meningeal mass lesion. METHODS This is a case report and review of literature. RESULTS A 38-year-old man developed thoracic back pain that progressed over one month with associated severe focal tenderness over his mid to lower thoracic spine. He developed intermittent numbness of the bilateral lower extremities, nuchal rigidity, difficulty sleeping, and night sweats. An MRI of the thoracic spine demonstrated a dorsal intradural, extramedullary enhancing lesion from T6-T9 extending outside the spinal canal. Dural thickening across the entire circumference of the spinal cord was noted. CT-guided biopsy of the thoracic lesion was performed, and pathology was consistent with follicular lymphoma. FDG PET: CT demonstrated no systemic disease. Bone marrow biopsy was negative for malignancy. Symptoms resolved with dexamethasone therapy. He was treated with bendamustine and rituximab with follow up PET: CT two months later demonstrating a complete response. DISCUSSION This case illustrates an unusual presentation of follicular lymphoma as a spinal meningeal mass lesion. Though rare, lymphoma should be included in the differential diagnosis of a dural mass lesion. Prompt diagnosis and initiation of treatment can lead to complete response and resolution of symptoms.

Cardiac magnetic resonance follow-up of COVID-19 vaccine associated acute myocarditis

Mass COVID-19 vaccination campaigns have helped impede the COVID-19 pandemic. In rare cases, some vaccines have led to vaccine associated myocarditis in a specific subset of the population, usually young males. Cardiac magnetic resonance (CMR) can reliably diagnose vaccine associated myocarditis, but follow-up data of CMR proven acute myocarditis is scarce. Nine patients with acute vaccine associated myocarditis underwent baseline and follow-up CMR examinations and were compared to baseline parameters at initial presentation and to a group of 20 healthy controls. CMR protocol included functional assessment, T1 and T2 mapping, T2 signal intensity ratio, strain feature tracking, and late gadolinium enhancement (LGE). Myocarditis patients (n = 9, aged 24 ± 6 years, 8 males) underwent CMR follow-up after an average of 5.8 ± 4.3 months. All patients showed a complete resolution of visual myocardial edema while also demonstrating a reduction in overall LGE extent from baseline to follow-up (4.2 ± 2.1 vs. 0.9 ± 0.8%, p < 0.001), although visual LGE was still noted in all patients. Left ventricular ejection fraction was normal at baseline and at follow-up (58 ± 6 vs. 62 ± 4%, p = 0.10) as well as compared to a healthy control group (60 ± 4%, p = 0.24). T1 (1024 ± 77 vs. 971 ± 34 ms, p = 0.05) and T2 relaxations times (57 ± 6 vs. 51 ± 3 ms, p = 0.03) normalized at follow-up. Most patients reported a resolution of clinical symptoms, while two (22%) reported new onset of exertional dyspnea. Patients with COVID-19 vaccine associated acute myocarditis showed a complete, uncomplicated resolution of myocardial inflammation on follow-up CMR, which was associated with a near complete resolution of symptoms. Minor, residual myocardial scarring was present on follow-up LGE imaging. The long-term implications of the remaining myocardial scar-tissue after vaccine associated myocarditis remain unknown warranting further studies.

Long-lasting Symptoms After an Acute COVID-19 Infection and Factors Associated With Their Resolution

Persistent symptoms after SARS-CoV-2 infection are an emerging public health problem. The duration of these symptoms remains poorly documented. To describe the temporal dynamics of persistent symptoms after SARS-CoV-2 infection and the factors associated with their resolution. This cross-sectional study involved 53 047 participants from 3 French adult population-based cohorts (CONSTANCES [Consultants des Centres d'Examens de Santé], E3N/E4N, and Nutrinet-Santé) who were included in a nationwide survey about SARS-CoV-2 infection. All participants were asked to complete self-administered questionnaires between April 1 and June 30, 2020. Variables included sociodemographic characteristics, comorbid conditions, COVID-19 diagnosis, and acute symptoms. Blood samples were obtained for serologic analysis between May 1 and November 30, 2020, from patients with SARS-CoV-2 infection defined as enzyme-linked immunosorbent assay immunoglobulin G antispike detection confirmed with a neutralization assay. A follow-up internet questionnaire was completed between June 1 and September 30, 2021, with details on persistent symptoms, their duration, and SARS-CoV-2 infection diagnosis by polymerase chain reaction. Persistent symptoms were defined as symptoms occurring during the acute infection and lasting 2 or more months. Survival models for interval-censored data were used to estimate symptom duration from the acute episode. Multivariable adjusted hazard ratios (HRs) were estimated for age, sex, and comorbid conditions. Factors associated with the resolution of symptoms were assessed. A total of 3972 participants (2531 women [63.7%; 95% CI, 62.2%-65.2%]; mean [SD] age, 50.9 [12.7] years) had been infected with SARS-CoV-2. Of these 3972 participants, 2647 (66.6% [95% CI, 65.1%-68.1%]) reported at least 1 symptom during the acute phase. Of these 2647 participants, 861 (32.5% [95% CI, 30.8%-34.3%]) reported at least 1 persistent symptom lasting 2 or more months after the acute phase. After 1 year of follow-up, the estimated proportion of individuals with complete symptom resolution was 89.9% (95% CI, 88.7%-90.9%) with acute symptoms. Older age (>60 years; HR, 0.78; 95% CI, 0.68-0.90), female sex (HR, 0.64; 95% CI, 0.58-0.70), history of cancer (HR, 0.61; 95% CI, 0.47-0.79), history of tobacco consumption (HR, 0.80; 95% CI, 0.73-0.88), high body mass index (≥30: HR, 0.75; 95% CI, 0.63-0.89), and high number of symptoms during the acute phase (>4; HR, 0.43; 95% CI, 0.39-0.48) were associated with a slower resolution of symptoms. In this cross-sectional study, persistent symptoms were still present in 10.1% of infected individuals at 1 year after SARS-CoV-2 infection. Given the high level of cumulative incidence of COVID-19, the absolute prevalent number of people with persistent symptoms is a public health concern.

Ertapenem-induced neurotoxicity in an end-stage renal disease patient on intermittent haemodialysis: a case report

BackgroundCarbapenem-induced neurotoxicity is an unusual side effect, with seizure being the most commonly reported symptom. Among the carbapenems, imipenem-cilastin is classically associated with the most severe neurotoxicity side effects. Carbapenem is mainly excreted by the kidney and its half-life is significantly increased in patients with chronic kidney disease (CKD). Therefore, dose adjustment is necessary in such patients. Ertapenem-associated neurotoxicity is increasingly being reported in CKD patients, but rarely seen in patients with recommended dose adjustment.Case presentationWe report a case of a 56-year-old male patient with chronic kidney disease 5 on dialysis(CKD 5D). The patient presented with a history of fever, chills and rigours during a session of haemodialysis (HD). He was diagnosed with Enterobacter cloacae catheter-related blood stream infection and was started on ertapenem. After 13 days of ertapenem, he experienced an acute confusional state and progressed to having auditory and visual hallucinations. His blood investigations and imaging results revealed no other alternative diagnosis. Hence a diagnosis of ertapenem-induced neurotoxicity was made. He had complete resolution of symptoms after 10 days’ discontinuation of ertapenem.ConclusionOur case draws attention to the risk of potentially serious toxicity of the central nervous system in HD patients who receive the current recommended dose of ertapenem. It also highlights that renal dosing in CKD 5D patients’ needs to be clinically studied to ensure antibiotic safety.

A novel treatment workflow of idiopathic hypereosinophilic syndrome: a single-center retrospective cohort study.

A novel treatment workflow of idiopathic hypereosinophilic syndrome: a single-center retrospective cohort study.

Arachnoid Granulation Causing Unilateral Pulsatile Tinnitus Treated With Dural Venous Sinus Stenting.

Large arachnoid granulations that protrude into dural venous sinuses and partially obstruct outflow are an underappreciated etiology of pulsatile tinnitus (PT). Endovascular dural venous sinus stenting is thought to diminish turbulent venous outflow and may relieve obstruction caused by arachnoid granulations. Four patients at two institutions were evaluated for unilateral PT. Magnetic resonance imaging and digital subtraction angiography revealed moderate-to-severe stenoses from large arachnoid granulations within the implicated transverse sinus. All patients underwent venous manometry and endovascular sinus stenting. All patients experienced immediate and complete remission of their PT. Stenoses were relieved by a mean of 93% by Warfarin-Aspirin Symptomatic Intracranial Disease criteria. There were no procedural or periprocedural complications. All patients continued to report complete symptom resolution at a mean of 8-month follow-up. PT from arachnoid granulations are an underappreciated pathomechanism. Endovascular dural venous sinus stenting is an effective intervention for treating unilateral PT secondary to large arachnoid granulation.

Effective Treatment of Chronic Cough with Tinidazole as the Newest Antiprotozoa against Lophomonas blattarum.

Chronic cough is a common problem in the setting of family physicians. Recently, Lophomonas blattarum was considered a cause of respiratory symptoms in children and adults. This study is aimed at determining the effect of antiprotozoal treatment of Lophomonas in patients with a chronic cough in Mashhad during 2020-2021. This study was a randomized clinical trial. In this study, 60 patients with chronic cough and unremarkable imaging findings, who were unresponsive to three steps of standard treatment, were randomly assigned to the treatment, with 2 weeks of tinidazole and placebo. The tinidazole and placebo were prepared in a completely identical shape, and a random assignment was performed by a third party. The primary outcome was a complete resolution of cough. A follow-up of treatment was performed. Data were analyzed using the SPSS software version 25. The basic demographic results showed no significant differences of sex and age between two groups. The results of this study showed a complete resolution of all respiratory symptoms in 40% (12), a complete improvement of cough in 40% (12), and a complete resolution of dyspnea in 50% (10) of the tinidazole group. The remaining showed significant improvement in the severity of cough and dyspnea. Postnasal drip, sputum, body temperature, and airway hyperresponsiveness were improved significantly. After tinidazole treatment, laboratory assessment of bronchial lavage and sputum revealed that 86 percent of smears were converted to negative. Tinidazole effectively resolved the chronic cough and most of the respiratory symptoms. Lophomonas blattarum is a potential mechanism for chronic cough.

Paternal Postpartum Depression

This is a case study of a 28-year-old man without former psychiatric history who presented to the emergency room for persistent thoughts of harming himself and his 2-month-old son for the past 1.5 months. He had also been experiencing anhedonia, feelings of guilt, decreased concentration, interest, sleep, and energy over the past month following the birth of his son. He was diagnosed with major depressive disorder and admitted to inpatient psychiatry ward to mitigate safety concerns. During his admission, his diagnosis was amended to postpartum depression based on onset of symptoms and association with the birth of his son. He was started on 10 mg vortioxetine and was discharged once suicidal and homicidal ideations resolved. He was provided outpatient services to include Cognitive Behavioral Therapy and continued medication management on vortioxetine. Outpatient treatment course resulted in complete resolution of symptoms. [ Psychiatr Ann . 2022;52(11):484–486.]

8445 Surgical Management of Urethral Prolapse: A Case Report

<h3>Study Objective</h3> To describe a case of acute urethral prolapse presenting with refractory pain and urinary retention, and to demonstrate surgical management, delineating key surgical steps using a surgical video. <h3>Design</h3> Case report demonstrated in a narrated surgical video <h3>Setting</h3> Patient presented to an academic hospital emergency department and required emergent surgical management in the operating room. In the operating room, patient is positioned in dorsal lithotomy with LoneStar retractor in place. <h3>Patients or Participants</h3> This surgical video describes a case report of a 45-year-old perimenopausal female who presented with acute onset refractory pain and urinary retention, as well as inability to tolerate catheterization, secondary to acute urethral prolapse with thrombosis and necrosis. <h3>Interventions</h3> After appropriate patient positioning, healthy urothelium was identified with cystourethroscopy above the prolapsed tissue. Next, normal appearing urothelium was tagged proximal to the prolapse at 3 o'clock, 6 o'clock and 9 o'clock using interrupted 4-0 Vicryl sutures so that this healthy tissue would not retract once the prolapse was excised. The base of the prolapsed portion of the urethral was injected with 1% lidocaine with epinephrine. The prolapsed portion was excised with Bovie electrocautery in quadrants around the urethra. A 16 French transurethral Foley catheter was placed, and the balloon inflated. Normal healthy appearing urothelium was re-approximated to the external urethral meatus epithelium using interrupted 4-0 Vicryl sutures with repair of one quadrant at a time. This was performed over the Foley catheter to ensure patency and prevent stricture. <h3>Measurements and Main Results</h3> The patient presented in this surgical video had immediate and complete resolution of symptoms. <h3>Conclusion</h3> Urethral prolapse rarely requires surgical intervention; however, when surgical treatment of urethral prolapse is necessary, it can be effective with appropriate surgical technique.

ODP526 Thyrotoxic Periodic Paralysis in Graves’ Disease: A rare presentation in an African American male

Abstract INTRODUCTION: Thyrotoxic Periodic Paralysis is a rare but life-threatening manifestation of hyperthyroidism characterized by hypokalemia, acute flaccid paralysis, and signs of hyperthyroidism. Despite being commonly seen in East Asian males, TPP has been increasingly reported in other parts of the world including western countries. Early recognition and prompt treatment of TPP is essential in preventing its grave and potentially life-threatening consequences. CASE PRESENTATIONA 21-year-old African American male patient with past medical history significant for hypertension and untreated hyperthyroidism presented to the emergency department with sudden onset bilateral lower extremity weakness and difficulty walking for one day. Upon arrival to the emergency department, vitals were unremarkable. On examination, patient had a decreased muscle tone in bilateral lower extremities with depressed deep tendon reflexes. The thyroid gland was diffusely enlarged. The remainder of physical exam was normal. Electrolyte abnormalities were also present with decreased serum potassium level of 1.2 mEq/L (3.6-5.1 mEq/L) and magnesium of 1.7 mg/dL (1.8-2.5 mg/dL). A low TSH level of &amp;lt;0. 010 mIU/mL (0.350-4.940 mIU/mL) with elevated free T4 levels of 4.30 ng/dL (0.70-1.48 ng/dL) were noted. CT Head/ Brain without contrast and Chest X ray were unremarkable. Potassium replacement was initiated with oral and intravenous potassium chloride. He was also started on oral atenolol (25 mg daily) and methimazole (10 mg daily). The patient's symptoms resolved in few hours after the treatment was started. An ultrasound of the thyroid gland showed diffuse heterogeneity of the gland most consistent with thyroiditis or similar abnormality. No definite focal abnormalities were noted. TSH receptor antibodies were also elevated with levels at 10.70 IU/L (≤1.75 IU/L) making Graves’ disease the most likely diagnosis. The patient's condition significantly improved along his hospitalization with complete resolution of symptoms and electrolyte abnormalities. He was subsequently discharged on atenolol and methimazole with outpatient follow up at the endocrinology clinic. DISCUSSION TPP is an underreported yet possibly fatal medical complication of hyperthyroidism. Prevalence is higher in Asia, however, incidence in Western countries has been increasing. While the underlying pathophysiology is poorly understood, h ypokalemia in TPP results from an intracellular shift of potassium induced by an increased adrenergic sensitization of Na + /K + –ATPase pump. Genetic and environmental factors may possibly play a role. Treatment of TPP entails beta-blockade to prevent intracellular shift of potassium, replenishing potassium stores, as well as correcting the underlying hyperthyroid state. Immediate diagnosis and initiation of treatment prevents serious cardiopulmonary complications and increases the probability of full recovery of muscle weakness. Presentation: No date and time listed

8144 Laparoscopic Robot-Assisted Hysterectomy with Vaginal Specimen Extraction Using Excite Technique of a Large Uterus

<h3>Study Objective</h3> Demonstrate a safe and didactic step-by-step hysterectomy of a leiomyoma large uterus using robotic platform and its vaginal specimen removal using Extracorporeal C-Incision Tissue Extraction (ExCITE) technique. <h3>Design</h3> Case report illustrated with a video. <h3>Setting</h3> Patient was placed in semi gynecological position, arms alongside the body and legs 80 grades abducted in adjustable leggings. Three 8mm robotic portals were placed on umbilical scar and both flanks. One additional 5mm portal was set in the right flank, for laparoscopic assistance. <h3>Patients or Participants</h3> 48-year-old woman reports abdominal bulge and pelvic pain for 6 months, with progressive worsening, radiating to lumbar region and legs. She also reports increased menstrual flow, with important loss of quality of life. MRI of the pelvis showed uterus with estimated volume of 920 cm³, heterogeneous myometrium due to multiple fibroids, the largest with 9.0 × 6.6 × 6.5 cm (vol 200 cm³), vascularized, determining compression on the upper wall of the bladder. <h3>Interventions</h3> Robot-assisted laparoscopic total hysterectomy was indicated. In the abdominal inventory, an 8x enlarged uterus was observed with multiple intramural and subserosal myomas, as well as thin adhesions between mesorectal and posterior uterine wall. Procedure was performed safely with practically no blood loss at all, and uterus was removed by vaginal access, with cold-knife morcellation, using ExCITE technique. <h3>Measurements and Main Results</h3> We obtained a safe and efficient surgical treatment on a large uterus hysterectomy using robotic surgery, with specimen removal by cold-knife morcellation. Patient evolved with complete resolution of symptoms and satisfactory postoperative recovery. <h3>Conclusion</h3> We showed with our case report a safe and reproducible step-by-step of a large uterus robotic hysterectomy, showing that robotic platform can be an important tool when dealing with challenging large uterus. Moreover, large specimen removal, which can also be tricky, can be easily and safely performed using ExCITE technique.

PSAT053 An Unusual Case of Central Adrenal Insufficiency Following the Diagnosis of Presumed Neuro-Sarcoidosis

Abstract Background Central adrenal insufficiency develops due to inadequate ACTH secretion by the pituitary gland (secondary) or decreased corticotropin-releasing hormone (CRH) secretion by the hypothalamus (tertiary).1 The hallmark of central adrenal insufficiency is inappropriately low levels of ACTH in the setting of low cortisol levels.1 Central adrenal insufficiency can be caused by CNS processes affecting the hypothalamus or pituitary gland, such as infection, autoimmune disease, malignancy, or the abrupt cessation of steroids.1 Here we present a rare case of central adrenal insufficiency following the diagnosis of presumed neuro-sarcoidosis. Clinical Case A 69-year-old female presents with fatigue, weight loss, and diffuse bilateral weakness. Vital signs were within normal with a physical exam significant for 3/5 strength in bilateral upper and lower extremities. MRI brain revealed leptomeningeal and pituitary stalk enhancement. CSF studies showed CSF lymphocytic pleocytosis and an elevated CSF ACE level 5.8 U/L. The most common etiologies of an elevated CSF ACE level are neuro-sarcoidosis or malignancy, and patient had no imaging findings of malignancy. With the elevated CSF ACE level along with imaging findings, she was given a presumptive diagnosis of neuro-sarcoidosis. She was treated with three days of IV methylprednisolone with mild improvement in symptoms. After discharge, she continued to have symptoms of diffuse weakness, fatigue, nausea and was re-admitted a few weeks later. Vitals on this admission revealed hypotension to 93/55, heart rate 66, SpO2 99%. Repeat MRI brain revealed resolution of leptomeningeal and pituitary enhancements. Due to the hypotension, morning cortisol levels were drawn and returned low at 0.4 ug/dL. ACTH stimulation test showed cortisol at 0 minutes low at 0.5 ug/dL and cortisol at 60 minutes at 9.7 ug/dL. ACTH prior to stimulation test returned low at 1.6 pg/mL. With reduced ACTH levels in the setting of low cortisol levels, the patient was diagnosed with central adrenal insufficiency. She began hydrocortisone treatment with complete resolution of symptoms. Further pituitary hormones were tested with TSH returning low at 0.04 uIU/mL; free T4 normal at 0.8 ng/dL; T3 low-normal at 39 ng/dL; FSH low for a post-menopausal female at 2.8 mIU/mL. This multi-hormonal insufficiency suggested the presence of an intracranial process contributing to hypothalamic-pituitary dysfunction. With initial neuro-imaging findings and elevated CSF ACE, the intracranial process causing her central adrenal insufficiency was presumed to be neuro-sarcoidosis. Due to resolution of symptoms on hydrocortisone therapy, definitive histologic evidence of neuro-sarcoidosis was not pursued. Conclusion This is a case demonstrating a rare presentation of central adrenal insufficiency following the diagnosis of presumed isolated neuro-sarcoidosis, with symptoms and radiographic findings responding rapidly to steroid therapy. References Nicolaides, N. C., Chrousos, G. P., &amp; Charmandari, E. Adrenal Insufficiency. [Updated 2017]. In: Feingold KR, Anawalt B, Boyce A, et al., editors Endotext [Internet]. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

Risk factors for peritumoral edema after radiosurgery for intracranial benign meningiomas: a long-term follow-up in a single institution.

Peritumoral edema (PTE) is recognized as a complication following stereotactic radiosurgery (SRS). The aim of this paper was to evaluate the risk of post-SRS PTE for intracranial benign meningiomas and determine the predictive factors. Between 2006 and 2021, 227 patients with 237 WHO grade I meningiomas were treated with Novalis linear accelerator SRS. All patients were treated with a single-fraction dose of 11-20 Gy (median 14 Gy). The median tumor volume was 3.32 cm3 (range 0.24-51.7 cm3). The median follow-up was 52 months (range 12-178 months). The actuarial local tumor control rates at 2, 5, and 10 years after SRS were 99.0%, 96.7%, and 86.3%, respectively. Twenty-seven (11.9%) patients developed new or worsened post-SRS PTE, with a median onset time of 5.2 months (range 1.2-50 months). Only 2 patients developed post-SRS PTE after 24 months. The authors evaluated factors related to new-onset or worsened PTE after SRS. In univariate analysis, initial tumor volume > 10 cm3 (p = 0.03), total marginal dose > 14 Gy (p < 0.001), preexisting edema (p < 0.0001), tumor location (p < 0.001), parasagittal location (p < 0.0001), superior sagittal sinus (SSS) involvement (p < 0.0001), and SSS invasion (p < 0.015) were found to be significant risk factors. In multivariate analysis, total marginal dose > 14 Gy (HR 3.38, 95% CI 1.37-8.33, p = 0.008), preexisting SRS edema (HR 12.86, 95% CI 1.09-4.15, p < 0.0001), tumor location (HR 2.13, 95% CI 1.04-3.72, p = 0.027), parasagittal location (HR 8.84, 95% CI 1.48-52.76, p = 0.017), and SSS invasion (HR 0.34, 95% CI 0.13-0.89, p = 0.027) were significant risk factors. Twelve (5.3%) patients were symptomatic. Ten of 27 patients had complete resolution of neurological symptoms and edema improvement with steroid treatment. Steroid treatment failed in 2 patients, who subsequently required resection for PTE. Radiosurgery is a safe and effective method of treating benign intracranial meningiomas according to long-term follow-up. We also identified total marginal dose > 14 Gy, preexisting PTE, parasagittal location, and SSS invasion as predictors of post-SRS PTE. Risk factors for post-SRS PTE should be considered in meningioma treatment.

PSAT302 Hypothyroidism Induced Nontraumatic Rhabdomyolysis: a Rare Cause of a Potentially Fatal Condition

Abstract Introduction Severe hypothyroidism can present with a vast array of clinical presentations, including musculoskeletal symptoms in up to 80% of patients. These symptoms can vary from simple muscle weakness and myalgias to rare complications such as rhabdomyolysis. Rhabdomyolysis, a syndrome characterized by degradation of muscle tissue, can occasionally lead to life-threatening complications including hyperkalemia, cardiac arrhythmias, and acute renal failure. We describe an interesting case of an otherwise healthy 19-year-old female with a past medical history of asthma who presented with nontraumatic rhabdomyolysis secondary to untreated hypothyroidism. Case description A 19-year-old female and college athlete of Asian descent with a past medical history of asthma presented to her primary care provider for acute and progressive swelling in her face, hands, and lower extremities bilaterally. In addition, she reported the onset of oligomenorrhea, decreased concentration, and myalgias for the past year. After relevant labwork was sent, the patient was found to have an extremely elevated TSH of 491.6 mIU/L with an undetectable level of free T4. She was referred for emergent hospital evaluation where notable lab findings included: creatinine 1.35 mg/dL, AST 300 IU/L, ALT 155 IU/L, LDH 888 IU/L, prolactin 41 ng/dL, and creatine kinase 8,380 IU/L. She was also found to be bradycardic (heart rate of 48 beats per minute) with low voltage QRS complexes on electrocardiography. She was admitted for treatment of newly diagnosed, severe hypothyroidism complicated by significant rhabdomyolysis. During her hospitalization, TPO antibodies were found to be negative however she was found to have a positive TSI level of 263% from baseline and positive antithyroglobulin of 6.7 units. Following treatment with weight-based intravenous levothyroxine (70 mcg daily), oral liothyronine (5 mcg twice daily), and intravenous fluid replacement with normal saline, the patient's condition and lab abnormalities improved significantly. She was eventually discharged and transitioned to oral levothyroxine (88 mcg). Seen in subsequent outpatient endocrinology follow-up, the patient has had normalization of all labs, including TSH, free T4, prolactin, creatinine kinase, and creatinine levels. She has experienced a complete resolution of myxedema symptoms and has had resumption of normal menses. Discussion Untreated or suboptimally treated hypothyroidism can be causative of various musculoskeletal manifestations including rhabdomyolysis. Hypothyroidism can induce muscle damage via various mechanisms including impaired glucose and triglyceride metabolism, glycosaminoglycan deposition in type I and II muscle fibers, decreased contractility of the actin-myosin unit and slowed ATP turnover. It is important to recognize hypothyroidism as a potential cause of non-traumatic rhabdomyolysis as prompt treatment with thyroid hormone can help prevent worsening muscle injury. As typically seen alongside myxedema with or without coma, treatment should be done in the inpatient setting with close hemodynamic and clinical monitoring and should include intravenous thyroid hormone, intravenous fluids and supportive care. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.