Documented Case Reports Research Articles

Cedecea lapagei as an emerging extensively drug-resistant microorganism: A case report in a patient with pleural empyema and literature review

Cedecea lapagei is a Gram-negative bacterium that belongs to the Enterobacteriaceae family and is said to be pathogenic for humans. Herein, we report a case of extensively drug-resistant C. lapagei in a patient with pleural empyema and offer a literature review of the already documented case reports on individuals infected with C. lapagei. A 60-year-old female patient was brought to the emergency department at AIIMS Bathinda with a history of breathing difficulty since one day after she sustained multiple injuries from a road accident. She was diagnosed with pleural empyema. Subsequently, an intercostal drain tube was inserted into the patient, and the drain content was sent for culture, which revealed growth of C. lapagei. Our literature retrieval work gathered a total of 13 relevant cases. In conclusion, early identification of C. lapagei and administering suitable treatment is important for good patient outcome.

The molecular basis of hypoprolactinaemia.

Hypoprolactinaemia is an endocrinopathy which is typically encountered as part of a combined pituitary hormone deficiency picture. The vast majority of genetic causes identified to date have been in the context of congenital hypopituitarism with multiple co-existent endocrinopathies. This is primarily with its closest hormonal relation, namely growth hormone. Acquired hypoprolactinaemia is generally rare in paediatric patients, and usually occurs together with other hormonal deficiencies. Congenital hypopituitarism occurs with an incidence of 1:4,000-10,000 cases and mutations in the following transcription factors account for the majority of documented genetic causes: PROP-1, POU1F1, LHX3/4 as well as documented case reports for a smaller subset of transcription factors and other molecules implicated in lactotroph development and prolactin secretion. Isolated prolactin deficiency has been described in a number of sporadic case reports in the literature, but no cases of mutations in the gene have been described to date. A range of genetic polymorphisms affecting multiple components of the prolactin signalling pathway have been identified in the literature, ranging from RNA spliceosome mutations (RNPC3) to loss of function mutations in IGSF-1. As paediatricians gain a greater understanding of the long-term ramifications of hypoprolactinaemia in terms of metabolic syndrome, type 2 diabetes mellitus and impaired fertility, the expectation is that clinicians will measure prolactin more frequently over time. Ultimately, we will encounter further reports of hypoprolactinaemia-related clinical presentations with further genetic mutations, in turn leading to a greater insight into the molecular basis of hypoprolactinaemia in terms of signalling pathways and downstream mediators. In the interim, the greatest untapped reserve of genetic causes remains within the phenotypic spectrum of congenital hypopituitarism.

Myxoid pleomorphic liposarcoma.

Myxoid pleomorphic liposarcoma (MPL) is an extremely rare adipocytic tumor, recently recognized as a distinct entity in the 5th edition of the World Health Organization (WHO) Classification of Soft Tissue and Bone Tumors. Predominantly found in the mediastinum of young women, MPLs exhibit a combination of histological features characteristic of myxoid liposarcoma and pleomorphic (lipo)sarcoma. Their unique molecular features distinguish MPLs from other liposarcomas. Unlike myxoid liposarcomas and well-differentiated/dedifferentiated liposarcomas, MPLs lack specific FUS/EWSR1::DDIT3 gene fusions and MDM2/CDK4 gene amplifications, respectively. MPLs are associated with complex karyotypes, further highlighting their distinct genetic profile. They demonstrate aggressive growth patterns, high recurrence rates, and a high tendency to metastasize. These factors contribute to a poor prognosis, with a median survival of approximately 22.6 months. The aim of this review article is to provide a comprehensive summary of previously documented case reports and studies related to MPLs. By shedding light on the intricate details of MPLs, researchers and clinicians can gain valuable insights that may pave the way for improvements in diagnosis, treatment, and patient outcomes in the future.

Irony of mandibular molar: A case series if management of radix

Endodontics is a comprehensive knowledge of the intricate nature of the root canal system is paramount. In case series we had investigates the intricate landscape of managing radix entomolaris, an additional root often encountered in mandibular molars. Success in endodontic treatment hinges on precise diagnosis, anatomical understanding, and meticulous procedural execution. The enigmatic nature of radix entomolaris introduces complexities due to variations in root canal morphology. Through meticulously documented case reports, we elucidate the diagnostic methods and successful endodontic procedures employed in two distinct patient cases presenting with symptomatic irreversible pulpitis.

Three Case Reports of Adult-onset Still Disease Associated with Breast Implantation.

Autoimmune syndrome induced by adjuvants (ASIA) is an uncommon clinical condition reported by Shoenfeld et al. Although this syndrome is not scientifically validated, numerous reports on it have been published, and the manifestations are postulated to be diverse, including generalized symptoms such as chronic fatigue, myalgia, arthralgia, or dry mouth, induced by exogenous substances, specifically adjuvants, which can encompass vaccines, organisms, and silicone. Concurrently, adult-onset Still disease (AOSD) is also an infrequent ailment, characterized by spiking fever, arthritis, skin rash, lymphadenopathy, and serositis. Although the precise pathogenesis remains incompletely understood, some case reports suggest that ASIA may be at the root of AOSD development with the same instigator. In this context, we present three cases of patients diagnosed with AOSD, which possibly could be considered an association with ASIA, years after undergoing breast reconstruction with silicone breast implants. In one case, the patient solely received medical treatment due to her refusal to have the implant removed, resulting in multiple flares and severe complications related to glucocorticoid therapy. Conversely, in the other two cases, a combination of immunosuppressive therapy and silicone breast implant explantation led to the complete resolution of clinical symptoms. To the best of our knowledge, there are only 10 documented case reports of AOSD associated with silicone breast implants insertion. We believe this report serves as a complementary addition to prior research and offers further insights into the ongoing debate about whether explantation should be carried out early in the clinical course or not.

Effect of Concurrent Administration of Herbs on the Pharmacokinetics of Drugs: A Review

Herbal medicines are currently in high demand, and their popularity is steadily increasing as an alternative medicine. This is as a result of their perceived effectiveness, fewer side effects and relatively low cost. They are being used simultaneously with therapeutic drugs for the treatment and management of numerous medical conditions, but due to the complex mixture of bioactive constituents they are capable of affecting the pharmacokinetics and pharmacodynamics of conventional drugs when administered concurrently. Of serious concern is the concurrent consumption of herbal products and conventional drugs. Herb–drug inter-action (HDI) is the single most important clinical consequence of this practice. Using a structured assessment procedure, the evidence of HDI presents with varying degree of clinical significance. While the potential for HDI for a number of herbal products is inferred from non-human studies, certain HDIs are well established through human studies and documented case reports. This herb-drug interactions (HDIs) may lead to modifications in plasma drug levels resulting in therapeutic failure of the drug or, alternatively, it may cause drug-induced toxicity. The main routes proposed for HDIs include cytochrome P450 (CYP450)-mediated inhibition or induction and transport and flow proteins. In our review, some herbal medicines used for the treatment of various diseases were highlighted and case reports of their pharmacokinetics and pharmacodynamics herb-drug interactions were analyzed. Therefore, this review can be a quick reference tool for physicians, pharmacists and other healthcare professionals involved in therapy, and counseling towards appropriate use of drugs to maximize clinical outcomes.

Alopecia and Periodontitis: Exploring the connecting dots.

Alopecia areata is a systemic disease with nonscarring hair loss from the scalp, face, or any part of the body. The disease with undetermined etiology and pathogenesis has a considerable impact on the social life of patients which gradually builds up stress and expedites further deterioration of their condition. Interestingly, in such patients, besides stress-relieving exercises and immunomodulators, well-timed dental assessment and prompt treatment have contributed to improving the prognosis of alopecia. This depicts an interrelationship between alopecia and oral foci of infections. Over the past few decades, periodontitis has remained to be one of the predominant forms of oral focus of infection for systemic diseases. The present review throws light on the role of stress and autoimmunity in establishing a possible correlation between alopecia and periodontitis. It is augmented with documented case reports, demanding a complete oral examination in patients with the unexplained origin of alopecia. An interdisciplinary approach is the need of the hour which can prevent the progression of both the diseases as well as other oral infections.

S2422 A Lichen for the Esophagus: A Rare Case of Esophageal Lichen Planus

Introduction: Lichen planus is an inflammatory condition of the skin and mucosal membranes. Sites of disease are variable and esophageal involvement is uncommon. We highlight a case of esophageal lichen planus masquerading as eosinophilic esophagitis. Case Description/Methods: A 59-year-old female with a past medical history of hypothyroidism status post thyroidectomy presents with progressive dysphagia and odynophagia to solids for 3 years. Symptoms initially began after thyroidectomy. Evaluation by ENT was notable for inflammatory changes of the oropharynx for which PPI trial was given for clinical features of silent reflux. Despite a 2-month course of therapy, symptoms persisted. Videofluoroscopic swallow study was unremarkable without evidence of aspiration or penetration. Subsequent EGD was notable for exudates, linear furrowing, and rings to suggest the diagnosis of eosinophilic esophagitis. Esophageal biopsy pathology features, however, showed band-like lymphocytic infiltration of the lamina propria and civatte bodies within the squamous epithelium consistent with esophageal lichen planus. Unfortunately, the patient was lost to follow up and subsequent treatment was unable to be initiated. Discussion: Esophageal lichen planus is an exceedingly rare entity with less than 100 documented case reports. It is likely underdiagnosed given that it can mimic other esophageal entities, such as eosinophilic esophagitis as seen in our patient. As such, histologic examination is necessary to identify key features of esophageal lichen planus which include lymphocytic infiltrates of the lamina propria as well as civatte bodies, which represent necrotic keratinocytes. Esophageal lichen planus has a tendency to be chronic and require topical or systemic therapy with steroids. Stricture development will typically require dilatation. Clinicians should consider esophageal lichen planus as part of their differential for dysphagia especially given the potential for malignant transformation to squamous cell carcinoma, which has been reported in 1-2% of cases.

65-Year-Old Man With Weight Loss and Dyspnea on Exertion

65-Year-Old Man With Weight Loss and Dyspnea on Exertion

Aspergillus brain abscess presents as sinusitis in immunosuppressed and hyperglycemic patient

Introduction: There are currently very few documented case reports of Aspergillus brain abscesses in the setting of persistent sinusitis symptoms and comorbid conditions of Type II Diabetes Mellitus and steroid treatment. Many cases of intracranial aspergillosis are secondary to a disseminated invasive Aspergillus infection or primary pulmonary aspergillosis. This case illustrates the importance and vitality of considering the rare but potentially lethal diagnosis of Aspergillus fumigatus brain abscess in the differential of persistent sinusitis, even in the absence of systemic symptoms. Case Report: A 73-year-old female with risk factors of Type II Diabetes Mellitus (T2DM) and steroid treatment presented to the hospital with persistent sinusitis symptoms lasting three months. She subsequently was diagnosed with Aspergillus brain abscess after bicoronal bifrontal craniotomy with pericranial flap. The patient was treated with voriconazole and amphotericin B for 3–6 months post-resection and follow-up with neurosurgery to monitor abscess reduction. She was admitted five more times after initial diagnosis for sequelae related to the brain abscess. The initial abscess did decrease in size but then began to worsen. The patient unfortunately passed away six months after initial presentation and diagnosis. Conclusion: Few cases of invasive rhino-orbito-cerebral Aspergillus brain abscesses secondary to Aspergillus sinus infections have been documented. If missed it can be lethal. Therefore, when a patient presents with persistent sinusitis, in the relevant context, Aspergillus brain abscesses are an important differential diagnosis that warrant consideration.

Case report of a Saccharomyces cerevisiae lung parenchyma infection in an immunocompetent 64-year-old male with a Zenker diverticulum

Saccharomyces cerevisiae is a yeast microorganism known to inhabit the gut micro-biome. It is commonly used in the fermentation process of beer, wine, bread making, and is naturally found in soil, plants, and fruit. Saccharomyces boulardii (subtype of Saccharomyces) and cerevisiae are closely related and are commonly used as a probiotic and in treatment for diarrhea. In rare circumstances, Saccharomyces has been shown to cause infection in the immunocompromised and intensive care unit (ICU) patients, with antibiotic use, and central venous catheter use. This prompts us to present one of the only documented case reports of a lung parenchyma infection with Saccharomyces cerevisiae in a 64-year-old immunocompetent male with a past medical history of chronic obstructive pulmonary disorder (COPD), atrial fibrillation, atrioventricular (AV) nodal ablation, heart failure with preserved ejection fraction (HFrEF (post-status automated implantable cardioverter-defibrillator)), coronary artery bypass graft (CABG), gastric esophageal reflux disease (GERD), and a Zenker diverticulum.

EP459: Rare clinical gene variant of SOX9: Acampomelic campomelic dysplasia

eP459: Rare clinical gene variant of SOX9: Acampomelic campomelic dysplasia

MYO5B mutation causing cholestasis in the absence of Microvillous Inclusion Disease: a rare case report

Background and Aim: Gene mutations known to cause Progressive Familial Intrahepatic Cholestasis (PFIC) 1, 2 and 3 have been long known. Newer variants, labelled PFIC 4, 5 and 6 have been described in recent literature. Myosin 5B (Myo5B) mutation is known to cause Microvillous Inclusion Disease (MVID) and has recently also been implicated in PFIC 6, a rare entity with only few documented case reports. We describe one such case report of a young girl presenting with cholestasis with normal serum Gamma-Glutamyl Transferase (GGT) activity, without MVID, diagnosed to have PFIC 6.

Raising awareness of acute onset of swelling and lip paraesthesia in a teenage patient

We report a case of papillary renal cell carcinoma in a 19-year-old patient that manifested as ipsilateral numbness of the lower lip and swelling in the region of masseter insertion. Despite there being widespread metastatic disease at presentation, the diagnosis was delayed with false reassurance from a normal brain scan and the assumption that the symptoms were due to infection related to the lower wisdom tooth. Once the primary tumour site was established, the patient responded to the relatively new immunotherapy treatments for advanced renal cell carcinoma as advocated in the 2019 NICE guidance. There are no other documented case reports that discuss papillary renal carcinoma with metastatic spread to the mandible in teenagers. CPD/Clinical Relevance: This case highlights the importance of malignancy being a differential diagnosis of lip paraesthesia irrespective of age.

Complex Regional Pain Syndrome Type II After Cervical Interlaminar Epidural Injection

BACKGROUND: Complex regional pain syndrome (CRPS) is a pain condition associated with autonomic and inflammatory features and is characterized by pain that is disproportionate in magnitude to the typical pain after similar injuries. The pathophysiology of CRPS is poorly understood, and many events have been implicated as causative factors. CASE REPORT: There are 2 previously documented case reports of CRPS after epidural steroid injections (ESI). This case report details the development of CRPS symptoms in a patient after receiving a cervical ESI. The precipitating event could have been trauma to a nerve root, nerve root irritation from the injectate, or contrast media reaction. Treatment focused on physical therapy and early intervention with a stellate ganglion block. The patient had complete resolution of her symptoms after 10 months. CONCLUSIONS: Although rare, CRPS due to direct nerve root injury or nerve root irritation can develop after an ESI; early diagnosis and treatment may result in a better outcome. KEY WORDS: Complex regional pain syndrome, CRPS, epidural steroid injections, TFESI, ILESI

Rapid Review and Assessment of CBCT for Lung Opacity Changes Suggestive of COVID-19 Infection

Purpose: A state of emergency was declared in Ontario, Canada on March 17, 2020 as the coronavirus disease 2019 (COVID-19) was increasing in rate of transmission. As we learned about the presentation of COVID-19, several documented case reports showed detection during a patient's radiation therapy treatment. Ground glass opacities in the lung that become visible on CT/cone-beam CT (CBCT) may indicate early signs of COVID-19 infection in individuals who are otherwise asymptomatic or mildly asymptomatic.

Avascular Necrosis of the Talus After Subchondroplasty.

Subchondroplasty (SCP) is a relatively new procedure, developed in 2007 for the treatment of bone marrow lesions (BMLs), that has shown promising results in the knee through several different case series. The foot and ankle literature, however, is sparse, with only a few documented case reports or case series. At our institution, we have identified several patients with complications after this specific procedure. As a result, we report our case series of patients who developed talar avascular necrosis (AVN) after undergoing SCP. A retrospective review was performed of patients who underwent SCP for a talar BML at our facility or who were referred to our facility after undergoing SCP at an outside institution. Patients were included if they developed radiographic evidence of talar AVN after the procedure. Patient demographics, comorbidities, concomitant intraoperative procedures, complications, and subsequent interventions were reviewed. Seven patients were identified as having radiographic evidence of talar AVN after SCP. Average time interval was 23 months postoperative from index procedure to radiographic confirmation of AVN. Two of the index procedures were performed at our institution, whereas 5 of the index procedures were performed at outside institutions and referred for further management. Three patients had documented risk factors for AVN before the SCP procedure. All 7 patients were symptomatic from the AVN. We identified 7 patients who went on to develop talar AVN after having undergone SCP. Level IV, case series.

2,4-Dichlorophenoxyacetic Acid Poisoning Mimicking as Organophosphorus Poisoning: A Case Report

Different compounds such as organochlorines, pyrethroids, fungicides, 2,4-dichlorophenoxy (2,4-D) herbicides, mushrooms, opioids, cartap compounds, and amitraz compounds can mimic organophosphorus (OP) poisoning. Muscle fasciculation, pulmonary edema, convulsions, bradycardia, hypotension, and smell caused by pyrethroids, as well as neurological signs, seizures, pulmonary edema, and smell caused by organochlorines can mimic OP poisoning. Miosis, vomiting, coma, and hypotension caused by opioids; miosis, bradycardia, altered sensorium, respiratory depression, and hypotension caused by amitraz compounds; and vomiting, breathlessness, altered sensorium, hypotension, and seizures caused by cartap compounds can also mimic OP poisoning. Mushroom poisoning and few fungicide compounds are also known to mimic features of OP poisoning. Hyperglycemia and glycosuria are the key hallmarks of amitraz poisoning. 2,4-D compounds can also mimic most of the features of OP poisoning; however, rhabdomyolysis, coma, and hyper/hypotonia are key differentiating features. Allergic manifestation and greenish discoloration of the contacted skin are the differentiating features of cartap poisoning.Treating all agriculture-related poisoning with atropine without confirming the compound can lead to a therapeutic misadventure. Here, we discuss the case of a patient who was referred to our Emergency Department (ED) with an alleged history of an unknown poison ingestion which was managed with atropinization for suspected OP poisoning in an outside hospital. On probing the history, the actual compound was found to be a 2,4-D herbicide. Very few documented case reports of 2,4-D poisoning are available in the literature. Hyper/hypotonia, coma, and skeletal muscle damage are the key differentiating features of 2,4-D poisoning. Our patient had skeletal muscle damage (rhabdomyolysis), evidenced by raised creatine kinase-total and creatine kinase-muscle/brain. As there is no specific antidote, we treated the patient with urinary alkalinization and supportive care. The patient had a favorable outcome in the ED.

A case report of an aorto-pulmonary-venacaval fistula after penetrating cardiac injury.

BackgroundPenetrating cardiac injury (PCI) is an accepted burden in high violent crime areas. Traumatic intracardiac fistulae are however not that commonly detected on initial presentation, with most of these injuries being detected post-operatively or at routine follow-up. The literature is devoid of general principles around the pre-operative planning and intra-operative management in these cases, and thus warrant documented case reports by clinical units experienced in the management of these challenging clinical scenarios.Case summaryWe describe a case report of a 29-year-old male patient presenting to our Cardiothoracic Unit with an aorto-pulmonary-venacaval fistula after a traumatic PCI. We describe the clinical presentation, diagnostic challenges, and institutional experience in the operative management of this case.DiscussionThe patient was treated successfully with repair via sternotomy and femoral cardiopulmonary bypass with no neurological, pulmonary, or cardiac sequelae at early follow-up. The importance of selective pre-operative imaging in PCI, appropriate pre-operative surgical planning, and surgical experience in the management of these injuries is highlighted in this case presentation.

Real-time, interactive website for US-county-level COVID-19 event risk assessment.

Large events and gatherings, particularly those taking place indoors, have been linked to multitransmission events that have accelerated the pandemic spread of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). To provide real-time, geolocalized risk information, we developed an interactive online dashboard that estimates the risk that at least one individual with SARS-CoV-2 is present in gatherings of different sizes in the United States. The website combines documented case reports at the county level with ascertainment bias information obtained via population-wide serological surveys to estimate real-time circulating, per-capita infection rates. These rates are updated daily as a means to visualize the risk associated with gatherings, including county maps and state-level plots. The website provides data-driven information to help individuals and policy makers make prudent decisions (for example, increasing mask-wearing compliance and avoiding larger gatherings) that could help control the spread of SARS-CoV-2, particularly in hard-hit regions.