Published Case Reports Research Articles

Hospitalization and Emergency Department Encounters for COVID-19 After Paxlovid Treatment - California, December 2021-May 2022.

Nirmatrelvir/ritonavir (Paxlovid) is a combination protease inhibitor that blocks replication of SARS-CoV-2 (the virus that causes COVID-19) and has been shown to reduce the risk for hospitalization and death among patients with mild to moderate COVID-19 who are at risk for progression to severe disease* (1). In December 2021, the Food and Drug Administration (FDA) issued an Emergency Use Authorization (EUA) for early treatment with Paxlovid among persons with mild to moderate cases of COVID-19 who are at high risk for progression to severe disease (2). FDA and a small number of published case reports have documented recurrence of COVID-19 symptoms or a positive viral test result (COVID-19 rebound) 2-8 days after recovery or a negative SARS-CoV-2 test result among patients treated with Paxlovid (3-7); however, large-scale studies investigating severe illness after Paxlovid treatment are limited. This study used electronic health record (EHR) data from a large integrated health care system in California (Kaiser Permanente Southern California [KPSC]) to describe hospital admissions and emergency department (ED) encounters related to SARS-CoV-2 infections during the 5-15 days after pharmacy dispensation of a 5-day treatment course of Paxlovid. Among 5,287 persons aged ≥12 years who received Paxlovid during December 31, 2021-May 26, 2022, 73% had received ≥3 doses of COVID-19 vaccine†, and 8% were unvaccinated. During the 5-15 days after Paxlovid treatment was dispensed, six hospitalizations and 39 ED encounters considered to be related to SARS-CoV-2 infection were identified, representing <1% of all patients to whom Paxlovid treatment was dispensed during the study period. Among these 45 persons, 21 (47%) were aged ≥65 years, and 35 (78%) had at least one underlying medical condition§ (8). This study found that hospitalization or ED encounters for COVID-19 during the 5-15 days after Paxlovid treatment was dispensed for mild to moderate COVID-19 illness were rarely identified. When administered as an early-stage treatment, Paxlovid might prevent COVID-19-related hospitalization among persons with mild to moderate cases of COVID-19 who are at risk for progression to severe disease.

Transient ipsilateral breast edema following COVID-19 vaccination

While ipsilateral lymphadenopathy following COVID-19 vaccination is a relatively common finding on mammography and breast ultrasound transient ipsilateral breast edema is rare with only a few published case reports. Radiologists should be aware of this potential imaging appearance of the breast and axilla following COVID-19 vaccination given the imaging features of edema such as trabecular thickening, skin thickening, and increased echogenicity can overlap with benign and malignant breast conditions such as mastitis and inflammatory breast cancer. We describe a case of transient ipsilateral breast edema after COVID-19 vaccination followed by a discussion summarizing the current understanding and management of transient ipsilateral breast edema following COVID-19 vaccination.

The overall benefits of empagliflozin treatment in adult siblings with glycogen storage disease type Ib: one year experience.

IntroductionRecently published case reports suggest the benefit of empagliflozin use in subjects with glycogen storage disease Ib (GSD Ib).MethodsWe present the clinical and laboratory data of 2 adult brothers with GSD Ib treated with empagliflozin for 12 months.ResultsThere was no severe infection during administration of empagliflozin. The improvement of clinical symptoms of inflammatory bowel disease and arthritis along with reduction in serum CRP levels and urinary albumin excretion was noted. Neutrophil count increased, allowing for reduction or temporary withdrawal of G-CSF treatment.ConclusionsEmpagliflozin may be a new safe treatment in GSD Ib patients with an advanced stage of the disease.

A Scoping Review and Population Study Regarding Prevalence and Histopathology of Juvenile Vulvar Melanocytic Lesions. A Recommendation

Cases of vulvar melanocytic lesions in juveniles are rarely reported. We analyze the evidence regarding vulvar melanocytic lesions in juveniles with or without vulvar lichen sclerosus to help decision making by clinicians and pathologists. A scoping review on vulvar melanocytic lesions with or without vulvar lichen sclerosus, including malignant vulvar melanomas, in females up to age 18 years was performed. In addition, the histopathology records of the cohort of all such lesions in The Netherlands from 1991 through 2020 were investigated, and a structured analysis of tissue samples of the subset of cases with lichen sclerosus was performed. The literature study performed confirms that vulvar melanomas in juveniles are extremely rare and that published case reports are often disputed. In The Netherlands, there are no cases of malignant vulvar melanomas up to age 18 years recorded from 1991 through 2020. Atypical histopathological features are often found in biopsies of vulvar nevi in juveniles, especially with concomitant lichen sclerosus, confirming earlier case studies in the literature. We conclude that even with atypical findings, vulvar melanocytic lesions in juveniles have a benign course. To avoid unnecessary and possibly mutilating procedures, we advise referral to an expert center and adaption of existing guidelines for vulvar melanocytic lesions in juveniles.

Pleomorphic Adenoma of the Palate: A Rare Case in an Adolescent With Cleft lip and Palate.

Benign salivary gland tumors are rarely found in children and adolescents compared with adults. Pleomorphic adenomas (PAs), the most common benign salivary gland tumor, account for only 1% of all head and neck lesions and fewer than 5% of all salivary gland tumors in individuals under the age of 16 years. The data on palatal PA in the first 2 decades of life is confined to published case reports and case series. To date, there has never been a report of palatal PA in a patient with cleft lip and palate. Here we describe an adolescent female with bilateral cleft lip and palate with PA of the hard and soft palate who underwent wide local excision and reconstruction with a buccal fat pad and buccal myo-mucosal flap.

Burden of illness of aromatic L-amino acid decarboxylase deficiency: a survey of physicians in Southern Europe

Background Aromatic L-amino acid decarboxylase deficiency (AADCd) is an ultra-rare genetic neurometabolic disorder caused by mutations in the DDC gene. Objective This retrospective, noninterventional study was designed to describe the burden of AADCd including the associated healthcare resource utilization in Southern Europe. Methods Eleven clinicians completed a patient case study survey for patients with AADCd currently or previously under their care, followed by an interview with each clinician to assess healthcare resource utilization, patient characteristics, and symptoms. Results Clinicians provided data for 20 patients with AADCd, of whom 60% were male. All patients experienced movement disorders, 90% exhibited developmental delay, 85% reported sleeping problems, and 80% experienced gastrointestinal problems. The symptoms varied with disease severity. Patients with AADCd received care from more than 16 different specialists including both medical and paramedical healthcare professionals. Hospitalizations and visits to accident and emergency departments were also frequent. Conclusion In terms of symptoms and healthcare resource utilization, the burden of illness of AADCd is substantial. This study provides insights into several aspects of the disease that are difficult to ascertain from published case reports.

POSTERIOR PNEUMOTHORAX IN A PATIENT WITH COVID-19 ON ECMO: A CASE REPORT

TYPE: Case Report TOPIC: Chest Infections INTRODUCTION: Pneumothoraces are a known complication of SARS-CoV-2, but the reported rate varies between studies. Posterior pneumothoraces are extremely uncommon with few published case reports. CASE PRESENTATION: A 51-year-old woman presented with dyspnea, fever, pulmonary infiltrates, and positive SARS-CoV2 nasopharyngeal PCR. Her respiratory status deteriorated, she was intubated, and she ultimately required VV-ECMO cannulation. Her hospital course was complicated by DVT, pulmonary aspergillosis, E coli pneumonia, and left sided hydropneumothorax treated with chest tube drainage. On hospital day 56, the patient abruptly developed a vasopressor requirement with a rapid elevation in lactate. CXR identified a large left air space. POCUS of the chest was remarkable for A-line artifact and absent lung sliding of the left posterior chest. CT confirmed a posterior pneumothorax. A 14Fr small-bore chest tube was placed by IR with improvement in the size of the pneumothorax and hemodynamic status, shown on image C below. The patient’s course was further complicated by right-sided hydropneumothorax due to E. coli and E. faecalis empyema that was treated with 14Fr chest tube drainage. She gradually improved, was decannulated from VV-ECMO, and on hospital day 107 was discharged to an acute rehabilitation facility with all chest tubes removed. DISCUSSION: Here we describe a patient’s hospital course complicated by repeated pneumothoraces including a left posterior loculated pneumothorax causing tension physiology. CONCLUSIONS: Available literature suggests pneumothorax may occur in 11% of COVID-19 patients treated with ECMO. The proper management of these patients is still unclear and clinicians must take these complications into account when directing care. DISCLOSURE: Nothing to declare. KEYWORD: Pneumothorax

Lacrimal Gland Enlargement as an Extraintestinal Manifestation of Crohn Disease: A Case Report

A 29-year-old woman was referred for a 3-month history of recurrent fever, abdominal pain, and diarrhea. Abdominal computed tomography (CT) scans revealed diffuse intestinal wall thickening involving the transverse colon. When her abdominal pain recurred 1 month later, she noticed bilateral upper eyelid edema with an “S-shaped” contour. Lacrimal gland CT scans revealed enlarged lacrimal glands. Crohn disease sometimes presents with extraintestinal symptoms including ocular complications. Dacryoadenitis is rarely associated with ocular complications. Enteroscopy revealed a noncaseating granuloma in the small intestine, and Crohn disease was diagnosed. We report the characteristics of dacryoadenitis resulting from Crohn disease and review published case reports.

Moraxella osloensis Bacteremia and Pneumonia in a Seventeen-Year-Old Patient

Moraxella osloensis ( M . osloensis ) is a rare cause of bacteremia and pneumonia in pediatric patients. The published case reports of M. osloensis bacteremia are limited. Among children, there are only seven cases of invasive disease from M. osloensis identified, and specific treatment guidelines are not available. Our case adds to the limited literature on this rare bacterium and highlights the need to better define antimicrobial resistance patterns. Here we report a case of a 17-year-old previously healthy female admitted with M. osloensis bacteremia and pneumonia. The patient initially presented with clinical and radiographic findings consistent with community-acquired pneumonia and was sent home after an emergency department (ED) visit with oral antibiotic treatment. She was advised to return for admission after the blood culture obtained in the ED reported growth and was noted to have persistent symptoms despite antibiotic adherence. Treatment with ceftriaxone for 3 days intravenously followed by 7 days of oral cefdinir resulted in rapid clinical improvement and resolution of her infection. Due to its sparse prevalence in the literature, there is currently no standard antibiotic regimen for treatment of invasive infections with M. osloensis ; a variety of therapies ranging from 7 to 14 days in duration have been successfully employed. While Moraxella species (e.g., Moraxella catarrhalis ( M . catarrhalis )) are typically ?-lactamase producing, reports regarding ?-lactamase production by M . osloensis have been inconsistent. M . osloensis is a rare cause of invasive infection in adolescents. Given limited published data regarding antibiotic sensitivities of this specific organism, microbiologic sensitivities should be obtained if possible and response to therapy should be closely monitored. Int J Clin Pediatr. 2022;11(2):56-59 doi: https://doi.org/10.14740/ijcp483

Post-stroke mania

IntroductionApproximately one-third of stroke survivors develop poststroke depression. Post-stroke mania is relatively rare, with a prevalence of less than 2%. One review of case reports of late-onset mania in 2015 demonstrated that 51% of the patients had established vascular risk factors. In 28% of cases, the treatment of underlying organic cause contributed to successful remission of the manic episode.ObjectivesFor this review, we aimed to compile published case reports from the past 20 years to review late-onset mania as one of the neuropsychiatric outcomes of stroke and its management.Methodsliterature search on Pubmed, PsychInfo, and Embase utilizing keywords combinations: Bipolar, Manic, Mania, Secondary, Stroke, Poststroke, Post-stroke, Elderly, Old, Late onset, Late-onset, Lateonset, Hemisphere, Brain, Vascular, Infarction.ResultsAmong the 17 case reports, the age of onset of manic episode ranged from 47 to 86 with a mean of 67 years. Of the 17 cases, the right hemisphere was the most frequently affected (14/17, 82%), with cerebrovascular lesion involving the left hemisphere in 3 cases (17.6%).ConclusionsClinicians should consider mania secondary to an organic cause in patients presenting with focal or soft neurological signs or symptoms, manic episode with atypical symptoms such as visual or olfactory hallucinations, altered mental status, disorientation, impairment in memory or cognition, unusual age of onset for bipolar disorder, or unusual illness course such as single episode of mania or poor response to psychopharmacologic treatment. Some reviews suggest combination of mood stabilizers and second-generation antipsychotics. Benzodiazepines recommended as an adjunctive drug for acute management such as agitation, aggressive behavior or disinhibition.DisclosureNo significant relationships.

Paroxetine-induced galactorrhea

IntroductionAntidepressant-induced galactorrhea and increases in prolactin levels have been sporadically reported among SSRI-related side effects.ObjectivesCurrent rapport presents a case of 39 y.o. female who developed several adverse effects on paroxetine - including galactorrhoea - which improved on discontinuation of the drug.MethodsCase discussion of 39-year old woman who was treated with paroxetine for her panic disorder and developed galactorrhoea with hyperprolactinemia that resolved upon discontinuation of the drug. Additionally, authors performed the literature search using PubMed and Embase to review the similar cases and used PDSP Database to assess the latest pharmacodynamic (PD) properties of paroxetine and other SSRI’s.ResultsLiterature review (1966–2020) revealed 24 prior published case reports of SSRI-induced galactorrhea in users of paroxetine (n=4), escitalopram (n=4), sertraline (n=2), citalopram (n=2), fluoxetine (n=3), fluvoxamine (n=2) and other non-assessable reports (n=7). Elevated prolactin levels were mostly observed with paroxetine and escitalopram and rarely with fluoxetine, fluvoxamine and sertraline. PD-assessment showed the highest binding affinity of paroxetine and escitalopram to SERT (kPi = 0.07-0.2 and 0.8-1.1 nmol/L respectively) compared to other SSRI’s, in absence of other relevant PD-propertiesConclusionsIncreasing body of evidence shows that galactorrhea does occur among paroxetine female users. Pharmacodynamic mechanism of action is poorly understood but given the modern insights in relationship in serotonin and dopamine circuits, we suggest that strong SERT inhibitory properties of paroxetine might lead to a tonic suppressive influence on dopamine neurotransmission. This physiological link may explain an increase in prolactin levels through dopamine depletion in the tuberoinfundibular pathway.DisclosureNo significant relationships.

Metamizole as a Rare Cause of Drug-Induced Liver Injury.

Drug-induced liver injury (DILI) is a heterogenous entity with a wide range of pathogenetic mechanisms and clinical manifestations. DILI is a diagnosis of exclusion. Metamizole (dipyrone) is an analgesic increasingly used in Europe, but there is limited information on its adverse effects. We report the case of a 56-year-old man with acute fever, malaise and general deterioration. Onset of symptoms occurred 12 hours after intake of metamizole for shoulder pain. The patient’s medical history was remarkable for three episodes of an inflammatory syndrome with hepatitis of unknown aetiology during the previous 3 years. However, retrospective enquiry showed each episode was preceded by metamizole intake shortly before symptom onset. Relevant differential diagnoses such as infection, vasculitis, autoimmune or metabolic diseases were excluded. Liver biopsy was compatible with DILI. Discontinuation of metamizole led to rapid clinical improvement and normalization of liver transaminases.Metamizole is a very rare and poorly known cause of DILI with only a few published case reports in the literature. Careful medical history taking is important to identify the causative agent. Prompt recognition and discontinuation of the drug is crucial. Patients must be informed to avoid this medication in future.LEARNING POINTSMetamizole is a rare cause of drug-induced liver injury (DILI).Taking a systematic medical and drug history is crucial for diagnosing DILI.DILI is a diagnosis of exclusion.

Clinical characteristics, therapy and long-term outcome of short-coupled idiopathic ventricular fibrillation. A systematic review of published case reports with new extended long-term follow-up

Abstract Funding Acknowledgements Type of funding sources: None. Background Short-coupled idiopathic ventricular fibrillation (SC-IVF) is a rare, potentially lethal arrhythmia, initiated by SC-PVCs. Only small series have been published. Purpose To assess the clinical characteristics, therapies and long-term outcomes of patients (pts) with SC-IVF. Methods Data from all published case reports on SC-IVF/Polymorphic VT were collected. Inclusion criteria were normal cardiac work-up including during follow-up (FU), and documented SC-PVC-VF available for our analysis and ruling out early repolarization syndromes and CPVT at VF onset. Updated information was provided by 82% of authors contacted, with 22 additional ECG tracings provided. Results A total of 100 pts’ cases were identified, including 82 who met inclusion criteria. Mean FU increased from 2.75+3.6 years in the original publications to 10.5+7.9 years (p&amp;lt;0.0001). There were 42 (51%) males, aged 39.6+13.5 years at the time of SC-IVF diagnosis. Pts presented with syncope (40%), aborted cardiac arrest (ACA) (16%), ACA + arrhythmic storm (13%), arrhythmic storm (10%), ICD shocks (8%) or palpitations (1%). Prior history included syncope in 32 (39%) pts. Mean coupling interval of the SC-PVC was 293.6+50.7ms. Presumed sites of PVC origin were the RV, LV and RVOT in 54%, 21% and 5% of pts, respectively. A total of 69 (84%) pts had an ICD implanted before or after SC-IVF documentation. During long-term treatment with quinidine (n=11) or verapamil (n=32), SC-IVF did not recur in 82% and 50% of pts, respectively (p=0.08). Ablation was performed in 43 (92%) of the 47 pts in whom it was attempted. Ablation sites were the Purkinje RV (43%) or LV (23%), and the non-Purkinje RV (19%) or LV (2%). Acute and late success rates after a first ablation were 83% and 61.7%, respectively. A second ablation in 7 pts yielded a 74.5% total success rate. No difference was found in the success rate of quinidine compared with ablation procedures (p=0.6). During FU, 4 pts died including 2 from sudden cardiac death; both pts were treated with verapamil and refused ICD implantation or replacement. Conclusion SC-IVF mainly presents following syncope. Quinidine and ablation procedures have comparable long-term high efficacy in arrhythmia control. The commonest sites of successful ablation are the RV and LV Purkinje fibers.

PO-684-01 CLINICAL CHARACTERISTICS, THERAPY AND LONG-TERM OUTCOME OF SHORT-COUPLED IDIOPATHIC VENTRICULAR FIBRILLATION. A SYSTEMATIC REVIEW OF PUBLISHED CASE REPORTS WITH NEW EXTENDED LONG-TERM FOLLOW-UP

Short-coupled idiopathic ventricular fibrillation (SC-IVF) is a rare, potentially lethal arrhythmia, initiated by SC-PVCs. Only small series have been published. To assess the clinical characteristics, therapies and long-term outcomes of patients (pts) with SC-IVF. Data from all published case reports on SC-IVF/Polymorphic VT were collected. Inclusion criteria were normal cardiac work-up including during follow-up (FU), and documented SC-PVC-VF available for our analysis and ruling out early repolarization syndromes and CPVT at VF onset. Updated information was provided by 82% of authors contacted, with 22 additional ECG tracings provided. A total of 100 pts’ cases were identified, including 82 who met inclusion criteria. Mean FU increased from 2.75+3.6 years in the original publications to 10.5+7.9 years (p<0.0001). There were 42 (51%) males, aged 39.6+13.5 years at the time of SC-IVF diagnosis. Pts presented with syncope (40%), aborted cardiac arrest (ACA) (16%), ACA + arrhythmic storm (13%), arrhythmic storm (10%), ICD shocks (8%) or palpitations (1%). Prior history included syncope in 32 (39%) pts. Mean coupling interval of the SC-PVC was 293.6+50.7ms. Presumed sites of PVC origin were the RV, LV and RVOT in 54%, 21% and 5% of pts, respectively. A total of 69 (84%) pts had an ICD implanted before or after SC-IVF documentation. During long-term treatment with quinidine (n=11) or verapamil (n=32), SC-IVF did not recur in 82% and 50% of pts, respectively (p=0.08). Ablation was performed in 43 (92%) of the 47 pts in whom it was attempted. Ablation sites were the Purkinje RV (43%) or LV (23%), and the non-Purkinje RV (19%) or LV (2%). Acute and late success rates after a first ablation were 83% and 61.7%, respectively. A second ablation in 7 pts yielded a 74.5% total success rate. No difference was found in the success rate of quinidine compared with ablation procedures (p=0.6). During FU, 4 pts died including 2 from sudden cardiac death; both pts were treated with verapamil and refused ICD implantation. SC-IVF mainly presents following syncope. Quinidine and ablation procedures have comparable long-term high efficacy in arrhythmia control. The commonest sites of successful ablation are the RV and LV Purkinje fibers.

Buccal Mucosal Grafts as a Novel Treatment for the Repair of Rectovaginal Fistulas: Protocol for an Upcoming Prospective Single-Surgeon Case Series.

BackgroundRectovaginal fistulas (RVFs) are abnormal communications between the rectum/anus and the vagina. They are most frequently formed a result of obstetric injury and have deleterious effects on patients’ quality of life. Despite several treatment modalities, RVFs remain difficult problems to manage, and many patients fail multiple attempts at surgical repair. Buccal mucosal grafts (BMGs) may be a solution to this problem. A BMG is an oral mucosal tissue harvested from the inner cheek. There are 2 case reports that describe the successful use of BMGs in the repair of RVFs.ObjectiveOur objective is to validate these findings with a prospective case series while also addressing the key issues of indication, technical details, procedure safety, and short-term outcomes.MethodsA prospective single-surgeon case series will be undertaken at a university-affiliated academic tertiary care hospital in Calgary, Alberta (Canada). The estimated recruitment is between 3 and 5 patients. Patients will undergo surgical repair of their RVFs with an autologous BMG. Data on patient characteristics, fistula characteristics, and surgical variables will be collected and analyzed prospectively. The primary outcome is fistula closure. This study has been approved by the Conjoint Health Research Ethics Board at the University of Calgary (REB20-1123).ResultsTwo previous case reports have described the successful use of BMGs in the repair of RVFs. We have received ethics approval to attempt to validate these findings through a prospective case series.ConclusionsRVFs cause significant patient morbidity and are difficult problems to manage. Bolstered by the successful use of BMGs in urologic surgery and the previously published case reports demonstrating success in RVFs, we believe that BMGs may be a solution to RVFs.International Registered Report Identifier (IRRID)PRR1-10.2196/31003

CARE guidelines for case reports: explanation and elaboration document. Translation into Russian

BACKGROUND: Well-written and transparent case reports (1) reveal early signals of potential benefits, harms, and information on the use of resources; (2) provide information for clinical research and clinical practice guidelines, and (3) inform medical education. High-quality case reports are more likely when authors follow reporting guidelines. During 20112012, a group of clinicians, researchers, and journal editors developed recommendations for the accurate reporting of information in case reports that resulted in the CARE (CAse REport) Statement and Checklist. They were presented at the 2013 International Congress on Peer Review and Biomedical Publication, have been endorsed by multiple medical journals, and translated into nine languages.&#x0D; OBJECTIVES: This explanation and elaboration document has the objective to increase the use and dissemination of the CARE Checklist in writing and publishing case reports.&#x0D; ARTICLE DESIGN AND SETTING: Each item from the CARE Checklist is explained and accompanied by published examples. The explanations and examples in this document are designed to support the writing of high-quality case reports by authors and their critical appraisal by editors, peer reviewers, and readers.&#x0D; RESULTS AND CONCLUSION: This article and the 2013 CARE Statement and Checklist, available from the CARE website [www.care-statement.org] and the EQUATOR Network [www.equator-network.org], are resources for improving the completeness and transparency of case reports.&#x0D; SOURCE: This article is a translation of the original paper CARE guidelines for case reports: explanation and elaboration document in the Journal of Clinical Epidemiology (doi: 10.1016/j.jclinepi.2017.04.026), prepared under the permission of the copyright holder (Elsevier Inc.), with supervision from the Scientific Editor by Professor E.G. Starostina, MD, PhD (translator) (Moscow, Russia).

P144 Phalangeal microgeodic syndrome: is this very rare condition becoming more common?

Abstract Background/Aims Phalangeal microgeodic syndrome (PMS) is a rare condition with only a handful of published case reports. Most of these cases are recognised in childhood, typically in Japan. It presents with pain and dusky discolouration in the digits, distinct from Raynaud’s phenomenon in that the colour change is fixed and pain constant. Symptom onset is related to cold exposure with first presentation usually occurring in winter. Features on magnetic resonance imaging (MRI) are striking and characteristic, with a distinct pattern of bone marrow oedema. Methods We report five cases of MRI-proven PMS all identified in the last 12 months, with symptom onset during the winter. The patients were all female, aged between 25 and 79. One patient was already under the care of rheumatology with an established diagnosis of systemic lupus erythematosus (SLE), but the remainder were de novo referrals presenting specifically with pain and discolouration of the digits. One patient developed symptoms 2 days after receiving the first dose of Pfizer/Biontech RNA vaccine against COVID-19. One patient had to self-isolate following a COVID-19 contact and developed symptoms shortly thereafter, although there was no proven infection in the affected individual. Results Four out of five cases were managed conservatively, with advice on maintaining core and peripheral temperature and skin integrity. Without any specific therapy, these four cases improved spontaneously to coincide with increasing seasonal temperatures. The fifth case with existing SLE was treated with intravenous pamidronate and subsequently with a calcium channel blocker, with some improvement in symptoms. Conclusion In these authors’ experience, it would seem PMS is increasing in incidence. It is unclear if this relates to wider availability and image quality of MRI, clinician bias due to personal experience of more cases or a genuine increase. The winter 2020/21 was particularly cold with sub-zero conditions lasting well into May, which may have impacted on this flurry of new cases. We have identified one prior case report which describes PMS in a patient with existing connective tissue disease. Our patient with SLE appeared to have a more severe clinical presentation requiring additional medical therapy to control the symptoms compared with the four cases where PMS was a stand-alone condition. Dermatological manifestations of acute COVID-19 infection are well documented in children and include chilblain-like lesions and acrocyanosis. Any association between COVID-19 and PMS is purely hypothetical and would require more study to identify any true relationship. Disclosure L. Parker: None. M. Ahmad: None. S. Mukherjee: None. P. McAlinden: None.

P039 Septic arthritis caused by Mycoplasma hominis in a patient with systemic lupus erythematosus

Abstract Background/Aims Mycoplasma hominis is a commensal bacterium of the genitourinary tract which can be associated with infection in immunocompromised patients. Colonisation with M. hominis has been associated with reactive arthritis and septic arthritis. We describe a case of M. Hominis septic arthritis in a patient with SLE on maintenance immunosuppression. Methods A 27-year-old woman presented with a 2-week history of a swollen left elbow and fatigue which affected her ability to work. Otherwise, she was systemically well and afebrile. She had an established diagnosis of SLE since she was aged 8 years, and her SLE had included mucocutaneous, central nervous system, renal involvement, and haemolytic anaemia. Her SLE was controlled on azathioprine, hydroxychloroquine, and prednisolone 5mg daily. Clinical examination revealed a mildly warm, non-erythematous, tender and swollen left elbow with a 40° flexion deformity. Initial investigations showed raised CRP 41mg/L and ESR 32mm/hr. White blood cell and platelet count were normal. Procalcitonin was insignificant at &amp;lt; 0.1ng/mL. Complements and urine analysis were normal, and dsDNA was negative. X-ray of the left elbow showed joint effusion. Ultrasound revealed active synovitis, subcutaneous oedema and purulent material in the joint. The fluid aspirate was straw coloured. Microscopy showed inflammatory cell count of 4230 white blood cells/mm, with 91% neutrophils. She was treated as probable septic arthritis with intravenous (IV) flucloxacillin. After 48 hours on IV antibiotics, her CRP remained elevated, and she developed pyrexia. Blood and joint fluid cultures were negative. MRI scan confirmed active synovitis with features suggesting joint infection. On discussion with microbiology, oral doxycycline was added on day 3 to cover possible atypical organisms. After a further 4 days the swelling, range of movement, and pain in her elbow had significantly improved. She was discharged home with oral flucloxacillin and doxycycline. On day 16 M. hominis was identified from the Bacterial Identification Services Colindale, by partial sequencing of 16s rDNA from the joint aspirate. She was therefore advised to continue with oral doxycycline for a total of 6 weeks. Results After treatment, her CRP, ESR and range of movement returned to normal. Conclusion This case highlights the potential pathogenicity of M. hominis in immunocompromised patients. A literature review found 35 published case reports of septic arthritis by M. hominis, with two occurring in patients with SLE. Approximately 70% of the affected cases were in immunocompromised patients who had a poor response to empirical antibiotic therapy. However, identification of M. hominis on joint aspiration and subsequent treatment with appropriate antibiotics resulted in good outcomes for all published cases. Disclosure R.M. Hum: None. H. Iftikhar: None. E.Y. Tang: None. I. Bruce: None. P. Ho: None.

Reports of Adverse Events Associated with Use of Novel Psychoactive Substances, 2017-2020: A Review.

An important role of modern forensic and clinical toxicologists is to monitor the adverse events of novel psychoactive substances (NPS). Following a prior review from 2013 to 2016, this critical literature review analyzes and evaluates published case reports for NPS from January 2017 through December 2020. The primary objective of this study is to assist in the assessment and interpretation of these cases as well as provide references for confirmation methods. Chemistry, pharmacology, adverse events and user profiles (e.g., polypharmacy) for NPS are provided including case history, clinical symptoms, autopsy findings and analytical results. Literature reviews were performed in PubMed and Google Scholar for publications using search terms such as NPS specific names, general terms (e.g., ‘designer drugs’ and ‘novel psychoactive substances’), drug classes (e.g., ‘designer stimulants’) and outcome-based terms (e.g., ‘overdose’ and ‘death’). Government and website drug surveillance databases and abstracts published by professional forensic science organizations were also searched. Toxicological data and detailed case information were extracted, tabulated, analyzed and organized by drug category. Case reports included overdose fatalities (378 cases), clinical treatment and hospitalization (771 cases) and driving under the influence of drugs (170 cases) for a total of 1,319 cases providing details of adverse events associated with NPS. Confirmed adverse events with associated toxidromes of more than 60 NPS were reported including synthetic cannabinoid, NPS stimulant, NPS hallucinogen, NPS benzodiazepine and NPS opioid cases. Fifty of these NPS were reported for the first time in January 2017 through December 2020 as compared to the previous 4 years surveyed. This study provides insight and context of case findings described in the literature and in digital government surveillance databases and websites during a recent 4-year period. This review will increase the awareness of adverse events associated with NPS use to better characterize international emerging drug threats.

Methicillin-resistant Staphylococcus aureus enterocolitis in a 16-month-old boy: a case report

BackgroundMethicillin-resistant Staphylococcus aureus enterocolitis is a rare disease that typically affects immunocompromised adults. Most cases of pediatric enterocolitis are caused by Gram-negative bacteria, Gram-positive Clostridiodes difficile, or viruses. This is the first published case report of a toddler with methicillin-resistant Staphylococcus aureus enterocolitis.Case presentationA 16-month-old non-Hispanic White boy with no past medical or psychosocial history initially presented to the emergency room with abdominal pain and emesis. Past family history was pertinent only for his father having a history of constipation. He was diagnosed with intussusception and underwent successful contrast reduction on hospital day 0. The following day, the patient had recurrent symptoms and a repeat contrast enema showed no evidence of recurrent intussusception. A computed tomography scan was obtained, which was concerning for possible recurrence with compromised bowel. He was taken to the operating room for operative reduction and underwent an ileocecetomy with primary handsewn end-to-end anastomosis. His postoperative course was complicated by an anastomotic leak on hospital day 6 necessitating reoperation and creation of an end ileostomy with mucous fistula. He received intravenous metronidazole, ceftriaxone, and ceftazidime antibiotics during his hospital course. On postoperative day 12, the patient developed a sudden increase in ileostomy output, and stool cultures were obtained. His symptoms persisted despite diet modifications, stopping antibiotics, and initiating loperamide. Three days later, stool cultures resulted negative for Escherichia coli, Salmonella, Shigella, Campylobacter species, and Clostridiodes difficile but were positive for methicillin-resistant Staphylococcus aureus. The patient was started on a 10-day course of oral vancomycin and discharged home in good condition 4 days later. After 12 weeks, the patient underwent reversal of the ostomy and is doing well at the 1 month postoperative follow-up, now 5 months from his initial surgery.ConclusionsTo our knowledge, this is the first published report of a toddler being diagnosed with methicillin-resistant Staphylococcus aureus enterocolitis. Because methicillin-resistant Staphylococcus aureus enterocolitis is rare and has overlapping symptoms with more common gastrointestinal pathologies, it is often misdiagnosed. When a patient presents with diarrhea or high ostomy output along with fecal cultures negative for Clostridiodes difficile and other common pathogenic agents, methicillin-resistant Staphylococcus aureus should be considered.