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Refractory septic shock and multiple organ dysfunction syndrome (MODS) remain a critical challenge in intensive care because they are associated with high morbidity and mortality despite advanced care. This case report describes the successful management of a 74-year-old female patient admitted with coronavirus disease 2019 (COVID-19) infection complicated by severe acute respiratory distress syndrome, refractory septic shock and MODS, including acute kidney injury and gastrointestinal bleeding. The patient received three sequential interventions, including continuous renal replacement therapy with the Oxiris hemofilter (Baxter International Inc, Deerfield, IL, USA) for cytokine/endotoxin removal, integrated extracorporeal CO2 removal (ECCO2R) via a single vascular access, and adjunctive IgM/IgA-enriched intravenous immunoglobulin therapy. The combined interventions resulted in a 74% reduction in C-reactive protein (325 → 85 mg/l), a 92% reduction in procalcitonin (28.3 → 2.3 ng/ml), normalization of hypercapnia (PCO2 107 → 48 mmHg) within 24h, and successful vasopressor discontinuation within 12 days. The patient recovered completely without any sequelae. This case report demonstrates how coordinated extracorporeal organ support can produce beneficial effects in patients with refractory septic shock. The combined treatment provided renal, respiratory, and immunologic support through minimal vascular access and warrants further investigation in controlled trials. Oxiris hemofilter rapidly improved haemodynamics Initiation of Oxiris-enhanced continuous renal replacement therapy (CRRT) was associated with a 40% reduction in norepinephrine requirement within 48 hours and a marked decline in inflammatory markers (C-reactive protein, CRP 325 → 188 mg/l; procalcitonin, PCT 28.3 → 9.1 ng/ml).Integrated Oxiris/ECCO 2 R rescued severe hypercapnic failure: During rebound crisis (PCO2 107 mmHg; pH 7.01), integrated extracorporeal CO2 removal (ECCO2R) via the CRRT circuit enabled lung-protective ventilation and normalized PCO2 to ~45-50 mmHg within 24 hours.Adjunctive IgM/IgA-enriched immunoglobulin supported recoveryAdding IV IgM/IgA-enriched immunoglobulin (5 ml/kg/day for 5 days) during rebound hyperinflammation was associated with further biomarker reduction (CRP 305 → 85 mg/l; PCT 15.8 → 2.3 ng/ml) and complete vasopressor discontinuation by day 12, suggesting potential synergy of sequential multi-targeted support.
Medullary thyroid carcinoma (MTC) is a rare neuroendocrine tumour arising from parafollicular C cells, and serum calcitonin remains its most reliable biochemical marker. Procalcitonin (PCT), widely used in the evaluation of bacterial infections, is usually undetectable under non-infectious conditions. Persistent elevation of PCT in the absence of clinical or microbiological evidence of infection is uncommon and may represent an early clue to underlying endocrine malignancy. We report the case of a 48-year-old woman who presented with markedly elevated PCT levels (>20 ng/ml) detected during routine laboratory testing despite being asymptomatic and having normal inflammatory markers. Extensive infectious work-up was negative, and antibiotic therapy did not reduce PCT levels. Further endocrine evaluation revealed a calcitonin level >2000 ng/l, elevated carcinoembryonic antigen, and a suspicious thyroid nodule on ultrasonography. Fine-needle aspiration with calcitonin washout confirmed the diagnosis of MTC. The patient underwent total thyroidectomy with central neck dissection, and histopathology demonstrated a 4.5 cm sporadic MTC without nodal metastasis. Postoperative PCT and calcitonin levels declined markedly, supporting their parallel behaviour in relation to disease activity. This case highlights the clinical importance of considering MTC in patients with unexplained hyperprocalcitoninemia and supports the potential role of PCT as an adjunct biomarker in diagnostic evaluation and postoperative monitoring once infection has been excluded. Persistent elevation of procalcitonin in the absence of infection should raise suspicion for medullary thyroid carcinoma and other neuroendocrine malignancies.Procalcitonin may serve as a useful adjunct biomarker alongside calcitonin in the diagnostic evaluation and postoperative follow-up of medullary thyroid carcinoma.Awareness of non-infectious causes of hyperprocalcitoninemia can prevent unnecessary antimicrobial therapy and reduce diagnostic delay in internal medicine practice.
Nebivolol, a third-generation selective β1-adrenergic receptor antagonist possessing vasodilatory properties, is characterised by a favourable safety profile. However, isolated cases of drug-induced liver injury (DILI) associated with its use have been documented in the literature. We present a case of hepatotoxicity in a 58-year-old female patient with a history of arterial hypertension, obesity and impaired glucose tolerance. Eight weeks after initiating nebivolol therapy (2.5 mg daily), a significant elevation in liver enzymes was observed: alanine aminotransferase (134.5 U/l), aspartate aminotransferase (118.1 U/l) and alkaline phosphatase (182.1 U/l), compared to previously normal baseline values. After excluding other potential causes (viral hepatitis, alcohol consumption, biliary obstruction), nebivolol was discontinued. Subsequent clinical and laboratory improvement was noted: transaminase levels decreased significantly within two weeks, and biochemical parameters had almost completely normalised after two months. The Naranjo Adverse Drug Reaction Probability Scale score was 7, indicating a probable causal relationship. Subsequent replacement of nebivolol with metoprolol did not result in recurrent hepatic dysfunction. This case substantiates the potential for idiosyncratic mixed-type hepatotoxicity associated with nebivolol administration. Although rare, this report underscores the importance of clinical vigilance and the consideration of DILI in cases of unexplained liver enzyme elevations in patients receiving this agent. Monitoring of liver function during the initial months of therapy is advisable, particularly in patients with underlying comorbidities. Nebivolol, despite its favourable safety profile can, rarely, cause significant mixed-type (hepatocellular-cholestatic) liver injury.Early recognition of drug-induced liver injury and prompt discontinuation of the offending agent are crucial for complete recovery.Monitoring of liver enzymes during the initial weeks of nebivolol therapy should be considered, especially in patients with metabolic risk factors.
Necrotizing fasciitis (NF) is a rapidly progressive soft-tissue infection associated with high mortality even when recognized early. Empyema necessitans (EN) is an uncommon complication of pleural empyema in which purulent material extends beyond the thoracic cavity into surrounding soft tissues. Although classically linked to Mycobacterium tuberculosis and Actinomyces, EN due to Streptococcus anginosus is distinctly rare. In exceptional cases, extra thoracic spread of empyema can track along fascial planes and evolve into NF. A 36-year-old previously healthy woman, recently treated for bilateral empyema, re-presented in severe respiratory distress and septic shock. She was found to have a foul-smelling open thigh wound, widespread soft-tissue involvement, and laboratory evidence of severe infection. Computed tomography (CT) imaging demonstrated bilateral multiloculated empyema with chest-wall extension consistent with EN, NF of the right thigh, multiple large retroperitoneal abscesses, and emphysematous cystitis (EC). She required intubation, vasopressor support, broad-spectrum antibiotics, and multiple surgical debridements. Pleural cultures grew S. anginosus. Retroperitoneal abscesses were managed conservatively, and multidisciplinary care led to steady clinical improvement. After approximately 5 weeks, her empyema had resolved, and her thigh wounds healed adequately, allowing discharge on oral antibiotics. This case illustrates an exceptionally rare progression of S. anginosus empyema to EN with concurrent NF and widespread retroperitoneal involvement. Early recognition of atypical dissemination, aggressive surgical management, and coordinated multidisciplinary care are essential for survival in such complex presentations. Streptococcus anginosus, although a commensal organism, has strong abscess-forming potential and can cause disseminated, life-threatening infections even in immunocompetent adults.This case highlights the importance of optimal treatment of lung empyema. Partially treated lung empyema can lead to empyema necessitans, with pus tracking along fascial planes and causing multiple superficial and retroperitoneal abscesses.Early recognition, repeated surgical source control, and multidisciplinary collaboration are critical to survival in multifocal invasive infections.
Urinary ascites secondary to spontaneous rupture of the urinary bladder (SRUB) is a rare clinical entity presenting with non-specific symptoms, which poses a significant diagnostic challenge. A specific presentation involves pseudo-renal failure because of peritoneal reabsorption of urinary solutes. We present the case of a female in her sixties with recurrent severe abdominal pain and ascites, concomitated with apparent acute kidney injury. Her medical history was significant for Wertheim-Meigs hysterectomy and pelvic radiation for cervical cancer. She underwent multiple hospitalisations with extensive investigations and even exploratory surgeries, driven by misleading CT findings of diffuse bowel wall thickening and gross ascites. The definitive diagnosis was made by extensive review of the patients' medical records and research of similar cases in the literature, as also by paracentesis, revealing an ascites-to-serum creatinine ratio of 3.76. A computed tomography cystogram confirmed an intraperitoneal bladder rupture with contrast extravasation. The patient was successfully managed conservatively with intermittent self-catheterisation. In patients with urogenital comorbidities developing large-volume ascites without preceding trauma, urinary ascites should be considered an important differential diagnosis, typically defined by an ascites-to-serum creatinine ratio of >1. This case explores the diagnostic pitfalls of urinary ascites secondary to SRUB in a patient with late radiation cystitis focusing on the phenomenon of reverse auto peritoneal dialysis and a normal cystatin C level as key diagnostic clues. Urinary ascites by spontaneous bladder rupture is a rare but critical late complication of pelvic radiation therapy, often misdiagnosed by its non-specific symptoms.Urinary ascites drives the diffusion of urinary solutes back into the systemic circulation, mimicking acute kidney injury by reverse auto peritoneal dialysis.The following constellation of laboratory parameters can be found in uroperitoneum: high serum creatinine but normal serum cystatin C, high serum-ascites albumin gradient and a high ascites-to-serum creatinine ratio.
Listeria innocua is widely distributed in the natural environment and food products. Human infections caused by L. innocua are extremely rare, with only approximately 5 cases reported globally over the past three decades (1990-2025). We report a case of a 74-year-old female with a history of poorly controlled type 2 diabetes mellitus (HbA1c 9.88%) and hypertension, who was admitted with headache, low-grade fever, and progressive altered mental status over 5 days. Cerebrospinal fluid analysis revealed pleocytosis. Both blood cultures (two sets) and the cerebrospinal fluid culture were positive for L. innocua, identified by matrix-assisted laser desorption/ionization-time of flight mass spectrometry. Despite an epidemiological investigation, the source of infection could not be determined. Initial treatment with ceftriaxone for 3 days failed with no clinical improvement. The patient was successfully treated with ampicillin for 49 days after culture results and antibiotic susceptibility testing became available. Cerebrospinal fluid parameters were normalized, and the patient was discharged after a 49-day treatment. This is a rare case of bacteraemia with meningitis caused by L. innocua in an elderly diabetic patient. The case emphasizes the importance of awareness of the pathogenic potential of L. innocua in immunocompromised patients, and the need to add ampicillin to empiric antibiotic regimens for meningitis in high-risk groups (age >65, diabetes mellitus, cancer, or immunosuppression). Listeria innocua, although considered non-pathogenic in healthy individuals, can cause severe bacteraemia and meningitis in elderly patients with poorly controlled diabetes mellitus.Third-generation cephalosporins are not effective against all Listeria species; empiric treatment in patients >65 years with diabetes mellitus and meningitis must include ampicillin.Recent molecular studies have identified some atypical L. innocua strains carrying virulence genes, challenging the traditional view of the harmlessness of this species.
Insulinoma is a rare functional pancreatic neuroendocrine tumour and the most common cause of endogenous hyperinsulinaemic hypoglycaemia in adults. Diagnosis is frequently delayed due to non-specific and intermittent symptoms, and a high index of suspicion is needed. We report the case of a 40-year-old non-diabetic woman who presented with recurrent episodes of weakness shortly after initiation of a glucagon-like peptide-1 receptor agonist (GLP-1RA). Laboratory evaluation demonstrated inappropriately elevated insulin and C-peptide levels during documented hypoglycaemic events, consistent with endogenous hyperinsulinaemia. Autoimmune and exogenous causes were excluded. Imaging studies identified a 2 cm hypervascular pancreatic lesion, with functional imaging confirming somatostatin receptor avidity consistent with an insulin-secreting tumour. The patient was initially treated with diazoxide as a bridge to definitive management. She subsequently underwent laparoscopic distal pancreatectomy, with histopathology confirming a well-differentiated pancreatic neuroendocrine tumour (WHO Grade 1). Following surgery, hypoglycaemia resolved completely. This case highlights the potential of GLP-1 receptor agonist therapy to unmask an insulinoma. Many benign insulinomas exhibit abundant expression of GLP-1 receptors. While a high index of suspicion remains essential, awareness of the emerging association between the incretin-based therapies and unexplained hypoglycaemia may facilitate earlier diagnosis. Hypoglycaemia is a rare but significant clinical event, and thorough evaluation is mandatory.Widely used GLP-1RA medications are glucose-dependent and therefore rarely cause hypoglycaemia; occurrence should prompt clinical suspicion for an underlying cause.Benign insulinomas express high levels of GLP-1 receptors; consequently, GLP-1 RAs may induce otherwise unexplained hypoglycaemia and can be used as tracers to localise difficult to detect tumours.
Dexmedetomidine is a highly selective α2-adrenergic agonist widely used for sedation in the intensive care unit (ICU) and is generally considered well tolerated, with hypotension and bradycardia being the most commonly reported adverse effects. Rarely, dexmedetomidine has been associated with polyuria resembling a diabetes insipidus-like syndrome, particularly during prolonged infusions. We report the case of a 53-year-old male admitted to the ICU for severe alcohol withdrawal who developed profound polyuria during a 5-day dexmedetomidine infusion. Urine output progressively increased to 6.75 litres per day, prompting an extensive evaluation that excluded osmotic diuresis, infection, intracranial pathology, and nephrogenic diabetes insipidus. Laboratory studies demonstrated preserved renal function and normal serum electrolytes. The temporal association between dexmedetomidine initiation and polyuria onset, along with resolution following drug discontinuation, supported the diagnosis of dexmedetomidine-associated polyuria, with a Naranjo adverse drug reaction probability score of 8 indicating a probable association. This case highlights an underrecognized adverse effect of dexmedetomidine. It underscores the importance of monitoring urine output and electrolytes during prolonged infusions to facilitate early recognition, prevent unnecessary diagnostic testing, and avoid complications related to volume depletion and electrolyte imbalance. Dexmedetomidine can, in rare cases, cause profound polyuria mimicking central diabetes insipidus, particularly during prolonged infusions beyond the Food and Drug Administration -approved 24-hour duration.Early recognition of dexmedetomidine-associated polyuria can prevent unnecessary diagnostic evaluations, reduce the risk of volume depletion and electrolyte disturbances, and prompt timely discontinuation of the offending agent.Internists managing critically ill patients should closely monitor urine output and serum electrolytes during extended dexmedetomidine use, especially in patients with hepatic impairment or refractory delirium.
Ovarian collision tumours are rare entities defined by the coexistence of two histologically distinct neoplasms, most commonly reported within the same ovary, while bilateral involvement is extremely uncommon and often under-recognised. Endometriomas are common benign ovarian lesions in women of reproductive age and may be asymptomatic or incidentally detected, whereas mature cystic teratomas are the most frequent ovarian germ cell tumours and are often discovered unintentionally during imaging or surgery. We report the case of a 27-year-old woman in whom adnexal lesions were incidentally identified, with no significant gynaecologic symptoms at presentation. Pelvic magnetic resonance imaging (MRI) showed bilateral ovarian masses, and surgical exploration revealed distinct ovarian lesions involving both ovaries. Histopathological examination confirmed a mature cystic teratoma in one ovary and an endometrioma in the contralateral ovary. This unusual and incidental bilateral presentation highlights the limitations of preoperative imaging in detecting synchronous ovarian pathologies and underscores the importance of thorough intraoperative assessment and definitive histopathological evaluation. Increased awareness of this rare entity is essential to optimise surgical management, fertility preservation and follow-up. Ovarian collision tumours are rare pathological entities, and presentations involving histologically distinct tumours in different ovaries are extremely uncommon.This case adds to the limited literature by illustrating an atypical and largely incidental presentation with non-specific symptoms.Such variability in clinical and radiologic presentation underscores the diagnostic complexity of these tumours and the potential for under-recognition.Although advanced imaging, particularly MRI, may provide important diagnostic clues, definitive diagnosis relies on a comprehensive histopathological assessment.Awareness of this unusual presentation is crucial, as early recognition of bilateral but distinct ovarian tumours can influence surgical planning, fertility-preserving strategies and follow-up, ultimately improving individualised patient care.
Herpes zoster results from reactivation of varicella-zoster virus (VZV) and classically presents with a dermatomal vesicular rash. Disseminated or visceral involvement is uncommon and predominantly occurs in immunosuppressed individuals. Gastrointestinal manifestations, such as oesophageal or gastric ulceration, are rare and can lead to diagnostic delay due to their non-specific presentation. We report the case of 70-year-old man presenting with progressive dysphagia and weight loss over four weeks. The symptoms were preceded by dermatomal facial vesicular lesions consistent with herpes zoster, followed by the appearance of non-dermatomal cutaneous lesions. The patient had underlying type 2 diabetes mellitus and was geriatric, constituting functional immunosuppression. Upper gastrointestinal endoscopy revealed multiple ulcerations in the oesophagus and gastric antrum. Histopathology examination demonstrated viral cytopathic changes compatible with VZV infection, supporting a diagnosis of disseminated visceral varicella. Due to limited antiviral availability, the patient was managed with supportive therapy, systemic corticosteroids and symptomatic treatment, resulting in significant clinical improvement and resolution of dysphagia. This case highlights disseminated visceral varicella as an important but under-recognised cause of gastrointestinal ulcerations in immunosuppressed elderly patients. Establishing standardised reporting criteria would allow for more persistent comparison and analysis of this rare manifestation of varicella infection. Disseminated varicella-zoster virus infection may involve the gastrointestinal tract and present as dysphagia or gastric ulcers in immunosuppressed patients.Elderly patients with diabetes mellitus are at increased risk for atypical and visceral manifestations of varicella-zoster virus, and increased risk of atypical or visceral varicella-zoster manifestations when gastrointestinal symptoms accompany recent or evolving skin lesions.While antiviral therapy is generally recommended for disseminated or visceral varicella-zoster infection, clinical improvement may occur with supportive care in selected cases. Nonetheless, close monitoring is mandatory and antivirals should be promptly initiated if clinical worsening is observed.
VEXAS (vacuoles, E1-enzyme, X-linked, autoinflammation, and somatic) syndrome is a newly recognized autoinflammatory hematologic condition due to mutations in the UBA1 gene. This case demonstrates its clinical variability, such as neutrophilic dermatosis, pulmonary disease, and a rare presentation with acute kidney injury and glomerulonephritis. A 77-year-old man with a history of benign prostatic hyperplasia, osteoarthritis, and patent foramen ovale presented with fatigue, night sweats, weight loss, and recurrent rashes, diagnosed as neutrophilic dermatosis. Laboratory results were pancytopenia and nephrotic-range proteinuria, and imaging revealed mediastinal lymphadenopathy and pulmonary ground-glass opacities. Bone marrow biopsy showed hypercellularity with vacuolization of myeloid and erythroid precursors. Renal biopsy revealed proliferative necrotizing and crescentic glomerulonephritis. Genetic analysis confirmed the UBA1 p.Met41Val mutation, diagnosing VEXAS syndrome. Treatment with corticosteroids, tocilizumab, and mycophenolate mofetil initially improved the symptoms, but steroid dependence continued. Severe pancytopenia necessitated changing to azacitidine after a year, and the patient was assessed for stem cell transplantation. His course was additionally complicated by neutrophilic pulmonary alveolitis. VEXAS syndrome presents with recurring fever, arthritis, pulmonary and cutaneous involvement, and cytopenias. Diagnosis needs to be entertained in the context of cryptic cytopenias and antineutrophil cytoplasmic antibodies (ANCA)-negative vasculitis. Renal involvement is atypical, especially in ANCA negative cases. Treatment continues to remain difficult, with the initial first-line therapy as high-dose glucocorticoids, with the alternative options being Janus kinase inhibitors, interleukin-6 inhibitors, and stem cell transplant. The case highlights the value of identifying both characteristic and atypical presentations of VEXAS syndrome. Prompt genetic testing is essential in diagnosis, and management entails a multidisciplinary strategy. VEXAS (vacuoles, E1-enzyme, X-linked, autoinflammation, and somatic) syndrome should be considered in patients with unexplained cytopenias, systemic inflammation, or atypical vasculitis.VEXAS syndrome is caused by a mutation of the UBA1 p.Met41Val gene.VEXAS syndrome usually presents itself as glomerulonephritis in antineutrophil cytoplasmic antibodies (ANCA) positive patients. However, it can be rarely present in ANCA negative patients too.
Yasunari nodules are choroidal lesions characterized by bright, poorly demarcated multiple lesions detected through near-infrared reflectance imaging and are considered diagnostic for neurofibromatosis type 1 (NF1). We report a case of unilateral Yasunari-like nodules in a patient without clinical or genetic evidence of NF1. A 53-year-old male presented with a 7-day history of floaters in both eyes. Best-corrected visual acuity was 20/20 in the right eye and 20/25 in the left eye. Slit-lamp examination revealed bilateral multiple iris nevi. Both fundi appeared normal on colour photography. The right infrared image demonstrated multiple bright, relatively well-defined areas suggesting Yasunari nodules, whereas the left infrared image was normal. Green and blue autofluorescent images were unremarkable bilaterally. Spectral domain optical coherence tomography revealed poorly demarcated hyperreflective choroidal nodule-like changes in the right eye only. Physical examination revealed no signs of NF1. Next-generation deoxyribonucleic acid sequencing of blood and skin specimens was negative for NF1. This is the second reported case of unilateral Yasunari-like nodules in a patient without NF1. Despite extensive clinical and genetic evaluation, including testing for somatic mosaicism, all results were negative for NF1. The lesions were considered benign incidental findings and monitored conservatively. Although infrequent, lesions resembling Yasunari nodules may be observed in patients without NF1. Clinicians should be aware of this possibility when encountering similar imaging findings. Clinicians should be aware of the fact that, although the presence of choroidal nodules (Yasunari nodules) is pathognomonic for the diagnosis of neurofibromatosis type I, similar choroidal appearances may occasionally be observed in normal eyes on infrared imaging.
Human parvovirus B19 is a highly prevalent single-stranded deoxyribonucleic acid virus that infects a large proportion of the global population. It can involve multiple organ systems, leading to a broad spectrum of clinical manifestations. While most infections in immunocompetent individuals are mild and self-limiting, parvovirus B19 can occasionally cause severe and diverse complications. We present a case of a 44-year-old, immunocompetent woman, who presented acutely with a severe acute inflammatory response associated with hepatomegaly, oedema, and cardiac and renal involvement. Early suspicion of atypical infections including parvovirus B19 was considered given the multi-organ involvement and non-specific aetiology. Parvovirus B19 infection was confirmed alongside the development of the typical rash seen in acute infection and the patient improved with supportive therapy with full resolution. This case underscores the potential severity of parvovirus B19 infection. Early testing and a high index of suspicion is required to accurately diagnoses these patients and may improve clinical outcomes whilst the mainstay of management is supportive. A potential underlying pathophysiology is suggested in acute parvovirus B19 infection with multi-organ involvement highlighting the link between infection and cytokine-driven inflammation and endothelial dysfunction. Early testing for parvovirus B19 should be considered in immunocompetent adults with features of acute hepatitis of unknown aetiology, especially when there is early haematological and joint involvement.In patients with acute infection with parvovirus B19, cytokine-driven inflammation and endothelial dysfunction can result in extremis and multi-organ involvement.Supportive treatment and close monitoring is the mainstay of treatment for patients identified and early escalation to intensive care may be required.
Mesalazine is a cornerstone therapy for ulcerative colitis (UC). Although generally safe, it can cause rare but life-threatening cardiopulmonary complications such as pleuropericarditis. We report on a patient with a UC flare who presented with dyspnoea, where concurrent pericardial and pleural effusions were detected. The inflammatory response was likely suppressed by concomitant corticosteroid therapy, which masked the clinical manifestations of mesalazine-induced serositis and delayed diagnosis. Two years later, the patient re-presented with recurrent massive pericardial effusion causing tamponade physiology and pleural effusion. Pericardial fluid analysis revealed a haemorrhagic, neutrophil-dominant profile (95% polymorphonuclear leukocyte, mimicking bacterial infection rather than the typically described eosinophilic pattern. Extensive cultures were negative. Following the discontinuation of mesalazine, rapid clinical and radiologic improvement was observed, confirming drug-induced pleuropericarditis. This case highlights that mesalazine-associated pleuropericarditis can be obscured by corticosteroids, leading to diagnostic delays. Furthermore, sterile inflammatory reactions to mesalazine can present with atypical neutrophilic features resembling bacterial infection, necessitating a high index of suspicion in inflammatory bowel disease patients presenting with serositis. A high index of suspicion for drug-induced serositis: in patients with inflammatory bowel disease, pleuropericarditis should be evaluated as a potential side effect of mesalazine rather than assuming it is an extraintestinal manifestation, especially when symptoms recur upon drug re-exposure.Corticosteroids may mask drug toxicity: concomitant corticosteroid therapy can temporarily suppress the clinical symptoms and effusion of mesalazine-induced pleuropericarditis, leading to a 'wrong footing error' and delayed diagnosis once steroids are tapered.Atypical laboratory features: mesalazine-induced sterile inflammatory reactions can present with a neutrophil-dominant effusion profile (rather than the typical eosinophilic pattern), which may mislead the clinician towards an infectious aetiology.
Bacteraemia caused by Kluyvera georgiana is an exceedingly rare clinical entity. As of 2024, only one case had been reported in the world literature: a 67-year-old female patient with recurrent pancreatic cancer undergoing chemotherapy in Japan, in whom the portal of entry for the pathogen was the biliary tract following choledochojejunotomy. We report the second case worldwide and the first from Vietnam, occurring in a 34-year-old male with decompensated cryptogenic cirrhosis. On hospital day 10, the patient developed high-grade fever (39.5°C) with rigors. Two independent blood culture sets drawn from two separate peripheral venous sites both yielded K. georgiana, identified by matrix-assisted laser desorption/ionization time of flight mass spectrometry. Notably, the patient exhibited leukopenia (1.710 cells/μl) with only minimally elevated procalcitonin (0.08 ng/ml), reflecting a blunted inflammatory response characteristic of cirrhosis-associated immune dysfunction. Antimicrobial susceptibility testing demonstrated resistance to ampicillin-sulbactam, ciprofloxacin, cefotaxime, ceftazidime, and trimethoprim-sulfamethoxazole, a substantially broader resistance phenotype than reported in the first case. The patient was treated with intravenous imipenem/cilastatin for 14 days and achieved complete clinical recovery. This case expands the recognized at-risk population for K. georgiana bacteraemia to include patients with decompensated cirrhosis, underscores the critical diagnostic value of early blood cultures when inflammatory markers are unreliable, and highlights the necessity of comprehensive antimicrobial susceptibility testing given the potentially variable resistance phenotypes among K. georgiana strains. Kluyvera georgiana bacteraemia can occur in patients with decompensated cirrhosis via gut bacterial translocation, expanding the recognized at-risk population beyond oncologic or chemotherapy-immunosuppressed hosts.Conventional sepsis biomarkers are unreliable in cirrhosis-associated immune dysfunction attenuates both leukocyte and procalcitonin responses. Blood cultures must be drawn whenever clinical suspicion arises, regardless of inflammatory marker values.Antimicrobial susceptibility of K. georgiana is unpredictable. Resistance profiles may vary substantially between strains, mandating comprehensive susceptibility testing and precluding empirical de-escalation based on historical data.
Maturity-onset diabetes of the young (MODY) KCNJ11 is caused by pathogenic variants in the KATP channel of the pancreatic β-cell leading to altered insulin secretion. Bi-allelic loss-of-function variants lead to neonatal diabetes mellitus (DM) whereas heterozygous gain-of-function variants lead to congenital hyperinsulinism. Heterozygous loss-of-function variants, however, are suggested to cause a bimodal pattern with hyperinsulinaemic hypoglycaemia in early life (with an often-mild presentation) and progressing to glucose intolerance in early adulthood. We describe three family members with early-onset DM (or glucose intolerance) without clear features of type 1 or type 2 DM. A heterozygous likely pathogenic missense variant c.119G>A (p.(Gly40Asp)) was found in an autosomal dominant pattern. This variant was previously only linked to cases of congenital hyperinsulinism and had been described as likely pathogenic or pathogenic in patients with type 2 DM. To our knowledge, this is the first report linking the c.119G>A KCNJ11 variant to a clinical context of MODY. We have no evidence in our patients of clinically significant hypoglycaemia at young age. However, we still hypothesise - congruent with the latest evidence - that there might be a bimodal pattern in individuals with this variant. This report adds to the clinical and genetic heterogenicity of MODY and illustrates the complexity of early diagnosis of MODY. We describe another clinical phenotype, namely diabetes mellitus, associated with a pathogenic variant in the KCNJ11 gene that was previously only linked to congenital hyperinsulinism.An initial non-specific genetic variant for MODY always warrants an extensive clinical work-up, a thorough family history and segregation of the variant in family members.This case highlights the heterogeneous presentation of pathogenic variants in MODY genes.
Parsonage-Turner syndrome (PTS), also known as neuralgic amyotrophy, is an uncommon neurologic disorder characterised by acute onset upper extremity pain followed by muscle weakness, atrophy and paraesthesias. Severity of symptoms is variable and can involve any peripheral motor nerves of the brachial plexus. Its occurrence in the immediate postpartum period is exceedingly uncommon. A 31-year-old G1P1002 with a twin gestation underwent an emergency caesarean delivery for non-reassuring foetal status of twin B following a spontaneous vaginal delivery of twin A. Her pregnancy was complicated by A2 gestational diabetes. Shortly after surgery, the patient experienced right arm numbness, followed by two weeks of severe pain. Upon resolution of pain, she developed profound weakness in the posterior interosseous nerve (PIN) distribution of the right hand. Physical examination revealed loss of finger extension at the metacarpophalangeal joints, weak thumb extension and loss of wrist extension, with decreased sensation to light touch in the radial four digits. Wrist flexion was preserved. Electromyography demonstrated right posterior PIN neuropathy; specialised MR neurography identified multiple hourglass constrictions, pathognomonic for PTS. The patient was managed conservatively with analgesics, a trial of corticosteroids, electrical stimulation and occupational therapy. Eleven months after symptom onset, finger extension returned spontaneously. This case highlights the presentation of PTS in the immediate postpartum period following caesarean delivery. Recognition of characteristic clinical features and imaging findings is crucial for accurate diagnosis. Conservative management can result in full functional recovery, though symptom resolution may be delayed. Parsonage-Turner syndrome should be considered in postpartum patients presenting with acute severe upper extremity pain followed by weakness, as the condition may be misdiagnosed as cervical radiculopathy, peripheral nerve compression or carpal tunnel syndrome.Hourglass constriction lesions identified on MR neurography are considered pathognomonic for neuralgic amyotrophy, but the presence of these lesions does not necessarily mandate early surgical intervention.Spontaneous neurologic recovery may occur even in patients with hourglass constriction lesions, supporting the importance of individualised management and careful longitudinal reassessment before proceeding to operative treatment.
Dermatomyositis (DM) is an autoimmune inflammatory myopathy that may rarely be drug induced. Statin-induced dermatomyositis (DIDM) is uncommon, with only few atorvastatin-related cases described in the literature. We present a case of a 62-year-old Hispanic woman who developed progressive proximal muscle weakness, myalgias, and joint discomfort shortly after initiating atorvastatin therapy. Despite discontinuation of the statin, her symptoms persisted and were accompanied by new cutaneous lesions. Laboratory evaluation revealed marked elevation of muscle enzymes, and subsequent skin biopsy revealed lymphocytic perivascular and perifollicular infiltrated with dermal mucin. Autoimmune serologies were negative, and malignancy screening was unremarkable. Treatment with systemic corticosteroids and mycophenolate mofetil resulted in gradual improvement. This report underscores the diagnostic challenges in differentiating statin-induced rhabdomyolysis from inflammatory myopathy, highlights the potential for rapid onset of dermatomyositis after statin exposure, and expands the limited literature on DIDM in Hispanic patients. By sharing this case, we aim to raise awareness among clinicians of this rare but clinically significant association, emphasizing the need for early recognition, histopathology, and malignancy screening to optimize outcomes. Statin-induced dermatomyositis is a rare immune-mediated complication that may mimic rhabdomyolysis early in its presentation.Lack of clinical or biochemical improvement after statin withdrawal and intravenous hydration should raise suspicion for inflammatory myopathy.Cutaneous findings, seronegative myositis profile, and skin biopsy features are key to distinguishing dermatomyositis from necrotizing autoimmune myopathy.
Eosinophilia, defined as an absolute eosinophil count >500 cells/μl, is most commonly secondary to allergic diseases, infections, or drug reactions. While parasitic helminth infections are a well-recognized cause, protozoal infections are less frequently associated. Reports linking Giardia lamblia infection with eosinophilia remain rare, in particular in the context of severe eosinophilia. A 77-year-old woman presented with acute watery diarrhoea for 3 days without systemic symptoms. Laboratory evaluation revealed marked eosinophilia (4.5 g/l at initial presentation, which rose to 7.9 g/l). The initial diagnostic workup, including stool polymerase chain reaction for bacterial and viral pathogens, helminth serologies, celiac screening, and abdominal imaging, was all negative. However, endoscopic evaluation with biopsies demonstrated mild eosinophilic gastrointestinal infiltration, and stool antigen testing was positive for Giardia lamblia. No other cause of eosinophilia could be identified. Treatment with metronidazole resulted in rapid clinical improvement and progressive reduction of eosinophil counts, with complete normalization during follow-up. Additional investigations only revealed mild hepatopathy, most likely related to statin therapy. This case highlights Giardia lamblia as a potential cause of marked secondary eosinophilia and thus may have to be considered as a differential diagnosis in patients presenting with unexplained severe eosinophilia and gastrointestinal symptoms. Giardia lamblia as a possible cause of severe eosinophilia.
Acute intermittent porphyria (AIP) is a rare hereditary metabolic disorder in haem biosynthesis that presents with severe abdominal pain. Attacks may be triggered by caloric deprivation. Diagnostic delay is common and reflects non-specific clinical presentations, the rarity of the disease and cognitive biases in clinical decision-making. A 30-year-old woman with opioid and benzodiazepine use disorder presented with abdominal pain and constipation and was initially treated for opioid-induced constipation. Persistent symptoms and hyponatraemia prompted further evaluation, revealing markedly elevated urinary porphyrin precursors. She was diagnosed with AIP and treated with intravenous hemin and high-carbohydrate therapy. This case illustrates the diagnostic challenge of AIP when plausible alternative explanations such as opioid-induced constipation are present. Persistent symptoms and hyponatraemia due to SIADH served as key clues prompting diagnostic reassessment. Diagnostic overshadowing may have contributed to delayed recognition in the context of substance use disorder. Concurrent severe malnutrition with caloric deprivation likely triggered the attack via accumulation of neurotoxic haem precursors, underscoring the importance of diagnostic flexibility in atypical or non-resolving cases. This case underscores the need to consider AIP in patients with severe abdominal pain and atypical clinical courses. Acute intermittent porphyria (AIP) can present as opioid-induced coprostasis, increasing the risk of diagnostic anchoring and delay.Coexisting factors (opioid therapy, psychiatric comorbidity, malnutrition) may act simultaneously as triggers, mimics and sources of cognitive bias.Reconsidering the initial diagnosis in atypical or non-resolving cases is essential to counter cognitive bias and improve outcomes in rare but treatable conditions.