de intensivist

EP01

Benralizumab as a Last Resort Therapy for Near-Fatal Eosinophilic Asthma in the Intensive Care Unit: A Case Series

M.P.H. Bischoff1, R.P.J. Segers1

1 Department of Intensive Care, Maastricht University Medical Centre+, Maastricht, the Netherlands

Abstract teaser

Bij patiënten met near-fatal eosinophilic asthma (NFEA) op de intensive care kan de standaardtherapie falen. Dit artikel beschrijft de klinische ervaring met off-label benralizumab bij vier therapieresistente patiënten. Na toediening vertoonden zij significante verbetering, wat suggereert dat benralizumab een mogelijke ‘rescue therapie’ kan zijn bij astma op de IC.

Background

Near-fatal eosinophilic asthma (NFEA) remains a life-threatening condition that may require mechanical ventilation and extracorporeal support despite maximal guideline-based therapy [1]. While biological therapy targeting interleukin-5 (IL-5) receptors, such as benralizumab, has proven effective in reducing exacerbations and eosinophilic inflammation in outpatient settings [2], its role in the intensive care (ICU) management of NFEA has not been established. Aim of the present study was to describe the clinical course and outcome of ICU patients with therapy-resistant NFEA treated with off-label benralizumab.

Methods

We conducted a retrospective case series of four adult patients admitted to the ICU with NFEA between 2019 and 2023, all of whom were intubated and showed persistent eosinophilia despite maximal pharmacological therapy. Off-label benralizumab 30 mg was administered subcutaneously after multidisciplinary consensus and family consent following the failure of standard treatments. Clinical trajectories, ventilatory parameters, airway resistance, and radiographic findings were evaluated before and after treatment.

Results

All four patients demonstrated clinical improvement following benralizumab administration. This included progressive reduction in ventilatory pressures and airway resistance (see Figures 1 and 2), successful weaning from extracorporeal membrane oxygenation in one case, and eventual extubation in all. No adverse events were reported. Due to the small study population, statistical analysis was not performed and results cannot be generalized.

Conclusions

In this first reported ICU case series, benralizumab appeared to facilitate recovery in patients with life-threatening, therapy-resistant NFEA after failure of standard treatment. The findings underscore the importance of proper asthma phenotyping and indicate the potential role of biological therapies in critically ill asthmatic patients. However, further studies are needed to establish the safety and efficacy of IL-5 receptor blockade in this context. In conclusion, while not routinely applicable, benralizumab may be considered for life-threatening eosinophilic asthma cases in selected patients.

References

• Louie et al., “The Critically Ill Asthmatic-From ICU to Discharge,” Clinical Reviews in Allergy and Immunology 43, no. 1–2 (2012): 30–44.
• M. FitzGerald et al., “Benralizumab, an Anti-Interleukin-5 Receptor α Monoclonal Antibody, as Add-On Treatment for Patients With Severe, Uncontrolled, Eosinophilic Asthma (CALIMA): A Randomised, Double-Blind, Placebo-Controlled Phase 3 Trial,” Lancet 388, no. 10056 (2016): 2128–2141.

 

EP02

  Thrombocyte count is an independent prognostic factor for thrombosis in the critically ill

F.J. Brouwer1, S.C. Schuett Rivrud2, R. Eck2, E. Keus2

1 Department of Intensive Care, St. Antonius Hospital Sneek, Sneek, the Netherlands

2 Department of Intensive Care, University Medical Center Groningen, Groningen, the Netherlands

Abstract teaser

Kunnen leukocyten- en trombocyten aantallen het optreden van VTE (veneuze trombo-embolie) tijdens ziekenhuisopname na een intensive care opname voorspellen?

Het absolute aantal trombocyten (OR 1.06 [1.02-1.11]) is significant geassocieerd met VTE tijdens ziekenhuisopname na IC-opname

Het absolute aantal trombocyten is een onafhankelijke prognostische factor voor VTE bij kritisch zieke patiënten en zou gebruikt kunnen worden voor toekomstige prognostische modellen.

Background

Immunothrombosis is the physiological phenomenon in which the innate immunity recognizes pathogens and damaged cells and orchestrates thrombus formation to inhibit pathogen dissemination and promote host survival. However, much like conventional hemostasis, this process can become dysregulated and occur inappropriately, potentially resulting in venous thromboembolism (VTE). Neutrophils, monocytes, and platelets have been described as the main actors in immunothrombosis1. We aimed to explore the prognostic importance of absolute counts of neutrophils, monocytes, lymphocytes, and platelets, as well as the neutrophil-lymphocyte ratio (NLR), monocyte-lymphocyte ratio (MLR), and platelet-lymphocyte ratio (PLR) for the occurrence of in-hospital VTE after intensive care unit (ICU) admission.

Methods

We conducted all analyses in a discovery cohort (the Medical Information Mart for Intensive Care; MIMIC-IV) database and repeated them in a replication cohort Simple Intensive Care Study (SICS 1 and 2).The outcome of interest was objectively proven

in-hospital VTE after ICU admission observed in routine practice. VTE was defined as pulmonary embolism or lower extremity deep vein thrombosis. We used univariable and multivariable logistic regression analysis to evaluate the prognostic importance of our candidate prognostic factors for VTE.

Results

Results

The discovery and replication cohort included 26,543 and 2,008 patients, respectively. Upon multivariable analysis in the discovery cohort, the absolute neutrophil count (OR 1.15 [1.11-1.19]), the absolute monocyte count (OR 1.08 [1.04-1.12]), the absolute lymphocyte count (OR 0.954 [0.929-0.98]), the absolute platelet count (OR 1.11 [1.11-1.12]), the NLR (OR 1.1 [1.07-1.13]), the MLR (OR 1.1 [1.08-1.13]), and the PLR (OR 1.11 [1.07-1.15]) were all statistically significantly associated with the occurrence of in-hospital VTE following ICU admission. Upon multivariable analyses in the replication cohort, only the absolute platelet count (OR 1.06 [1.02-1.11]) was significantly associated with in-hospital VTE following ICU admission. The remaining candidate prognostic factors in the replication cohort contained too much missingness (  80%) for a proper assessment of their prognostic importance.

Conclusions

The absolute platelet count is an independent prognostic factor for VTE in critically ill patients and should be used to inform future prognostic models. Furthermore, while absolute counts of neutrophils, monocytes, lymphocytes, and the NLR, MLR, and PLR may also be independent prognostic factors, we were unable to replicate these findings in the current study.

References

Engelmann B, Massberg S. Thrombosis as an intravascular effector of innate immunity. Nature Reviews Immunology 2012 13:1 [Internet]. 2012 Dec 7

 

EP03

Organ donation in the Netherlands: Factors affecting donor numbers in 2025

Nichon Jansen1, Aline Hemke1, Martin Heemskerk1, Nathalie van Dijk2, Walther van Mook2

• Nederlandse Transplantatie Stichting, Beleid & Onderzoek, Leiden, the Netherlands
• Department of Intensive Care, Maastricht University Medical Centre+, Maastricht, the Netherlands

Abstract teaser

Sinds de implementatie van het opt-out toestemmingssysteem in 2020 is het aantal orgaandonoren jaarlijks gestegen. Echter, in 2025 is een daling zichtbaar ten opzichte van 2024, waarbij de vraag is wat daarvan de oorzaak is. Analyse van data laat zien dat zowel het potentieel aan orgaandonoren op de IC’s als het toestemmingspercentage voor donatie zijn gedaald dit jaar.

Background

After the implementation of the opt-out consent system in 2020, a yearly increase in the number of organ donors was observed. However, lower numbers of donors were observed in 2025 compared to 2024. This study aimed to identify the factors of influence.

Methods

National data on deceased intensive care patients, collected by the Dutch Transplant Foundation through the application NovaNORD, were used to examine organ donor numbers. This study covers the period from January to October for the years 2021-2025.

Results

The annual number of organ donors increased between 2021 and 2024, from 271 to 285, 292, and 360, respectively; the current 2025 trend (249 donors as of October) is lower than that of 2024, see Figure 1. There are several possible explanations for the decrease in donor numbers in 2025 compared to the upward trend observed up to 2024. During the study period (January - October 2021 - 2025), the number of deaths in the ICUs initially declined from 6300 in 2021 to 5621 in 2022, but stabilized in the following years, reaching 5,559 in 2025, see Table 1. However, the number of potential organ donors rose steadily from 819 in 2021 to 937, 1,002, and 1,085 in 2024, returning to a level comparable to that of 2023 in 2025 (1,017).

The overall family consent rate fluctuated from 66% in 2021 to 62% in 2022, 64% in both 2023 and 2024, and declined to 62% in 2025. When families were approached in cases where the Donor Register indicated consent (Yes’ registration), the approval rate remained stable at approximately 86%. In contrast, when families were approached in cases of consent based on a ‘No objection’ registration, the approval rate declined from 48% in 2021 to 41% in 2025. For registrations indicating ‘Decision by next of kin/specific person,’ the consent rate decreased from 39% in 2021 to 28% in 2025.

Conclusions

In conclusion, the number of organ donors in 2025 is lower compared to 2024. However, it resembles the numbers from 2023. The main factors of influence appear to be a lower number of potential organ donors in ICUs in 2025, as well as a decline in the family consent rate for organ donation in cases of a ‘No objection’ and ‘Decision by next of kin/specific person’ registrations.

References

Dutch Transplant Foundation. Annual Reports 2021–2024. Leiden, The Netherlands; 2022–2025.

EP04

Continuous veno-venous hemofiltration for patients with metabolic acidosis in septic AKI: a Retrospective study

H.M. van der Weide1, J.W. Vermeijden1, M. Bombeld-Arkink1

1 Medisch Spectrum Twente, Intensive Care Centre, Enschede, the Netherlands

Abstract teaser

Het herstellen van een metabole acidose bij septische acute nierinsufficiëntie vraagt om een effectieve behandeling, dit kan doormiddel van niervervangende therapie. Wij vergeleken continue veno-veneuze hemofiltratie (CVVH) met continue veno-veneuze hemodiafiltratie (CVVHDF), in hun effect op bicarbonaat binnen 24 uur. CVVH corrigeert bicarbonaat effectiever en kan uitkomst bieden in het optimaliseren van de zorg van kritisch zieke patiënten.

Background

Metabolic acidosis is common in critically ill patients with sepsis and acute kidney injury (AKI). Continuous renal replacement therapy (CRRT) is often used to correct acid–base disturbances, with effectiveness depending on clearance mechanisms, bicarbonate concentration in replacement fluids, treatment dose, and the anticoagulation used. (1) In the study hospital, continuous veno-venous hemodiafiltration (CVVHDF) was the standard CRRT modality until November 2024, after which a new protocol was implemented introducing continuous veno-venous hemofiltration (CVVH) for patients with metabolic acidosis and sepsis. The aim of the present study is to compare CVVH and CVVHDF on increasing serum bicarbonate in patients with septic AKI and metabolic acidosis within 24 hours of treatment.

Methods

This single-center retrospective study included ICU patients with metabolic acidosis (pH ≤7.35, bicarbonate ≤20 mmol/L), sepsis, and AKI or acute-on-chronic kidney injury. Patients receiving CVVH with Regional Citrate Anticoagulant (RCA) and Phoxillium® (30 mmol/L bicarbonate) or CVVHDF with RCA and Biphozyl® (22 mmol/L bicarbonate) between 01-01-2024 and 30-06-2025, were included. The primary outcome was the difference in serum bicarbonate increase between both therapies within the first 24 hours.

Results

Twenty-two patients were included, seven received CVVH and fifteen CVVHDF. The increase in serum bicarbonate over 24 hours was significantly greater in patients receiving CVVH (p=0.002). (Figure 1)

The mean increase in bicarbonate was 9.1 (±4.2) mmol/L for CVVH (p<0.001) and 3.2 (±4.2) mmol/L for CVVHDF (p=0.002). The mean difference at 24 hours between the groups was 6.0 mmol/L (95% CI [2.2, 9.8], p=0.001).

CVVH patients were more likely to achieve normal serum bicarbonate (≥22 mmol/L) within 48 hours (hazard ratio=8.2) (Figure 2), but also had a higher risk of developing metabolic alkalosis (relative risk=1.75).

Conclusions

CVVH, with a high concentration bicarbonate buffer and RCA, is more effective than CVVHDF, with a lower concentration bicarbonate buffer and RCA, in increasing serum bicarbonate in the acute phase of the treatment of metabolic acidosis in patients with septic AKI. CVVH should be considered for such patients, with close monitoring for metabolic alkalosis.

References

1. Bouchard J et al. Acid-Base Disturbances in the Intensive Care Unit: Current Issues and the Use of Continuous Renal Replacement Therapy as a Customized Treatment Tool. Int J Artif Organs. 2008;31(1):6–14

References

• Bouchard J et al. Acid-Base Disturbances in the Intensive Care Unit: Current Issues and the Use of Continuous Renal Replacement Therapy as a Customized Treatment Tool. Int J Artif Organs. 2008;31(1):6–14

EP05

An unusual presentation of Goodpasture disease, avoid delay while getting the right diagnosis

C.M. van Deuzen1, A.F.C. Schut1, W. van den Tempel1

1 Department of Intensive Care, Ikazia Hostpital, Rotterdam, the Netherlands

Teaser Case report

Anti-glomerulaire basaal membraan (Goodpasture) ziekte is een kleine vaten vasculitis, die glomerulaire en pulmonale capillairen aantast; geïsoleerde pulmonale betrokkenheid bij patiënten >60 jaar is zeldzaam. Bij een patiënt met een “non-responding pneumonie” zonder hemoptoë bleek anti-GBM sterk verhoogd zonder renale betrokkenheid. Door spoedige behandeling middels methylprednisolon, cyclofosfamide en plasmaferese werd progressief orgaanfalen voorkomen. Wees alert bij atypische presentatie.

Case report

Introduction: Anti-glomerular basement membrane (anti-GBM) disease is rare with an incidence of 2:1.000.000, caused by circulating antibodies directed against GBM present in glomeruli and alveoli. Most patients present with rapidly progressive glomerulonephritis; in rare cases pulmonary disease predominates. Immediate start of treatment with steroids, immunosuppressive agents and plasmapheresis is essential to prevent further pulmonary failure and irreversible kidney damage.

Patient information: a 60-year-old man known with chronic obstructive pulmonary disease presented with dyspnoea three times in three weeks. During his first presentation pulmonary embolism was ruled out and he was sent home. The second time he presented with coughing white mucus and fever; antibiotics (cefuroxime followed by amoxicillin-clavulanic acid), prednisone and nebulizing resulted in clinical improvement. However, soon after discharge he was readmitted with worsening symptoms.

Clinical findings: Physical examination: respiratory rate 20/min, oxygen saturation 83% on ambient air, pulmonary wheezing and temperature 39.3°C. Laboratory results: CRP 237 mg/L (reference <10 mg/L), creatinine 70 micromole/L (reference 65-110 micromole/L) and normal urine analysis. Chest-CT: consolidations and ground-glass opacifications. Findings were most suitable with pulmonary infection with organising component.

Timeline: Patient was admitted to ICU and started on ceftriaxone and ciprofloxacine. Prednisone was continued. He was intubated at day three of admission due to progressive respiratory distress despite High Flow Nasal Oxygen. Bronchoscopy showed no endobronchial abnormalities. No micro-organisms were cultured. Additional workup to rule out auto-immune aetiology showed high anti-GBM IgG levels (552.7 kU/L (reference <20 kU/L)).

Therapeutic intervention and follow-up: Methylprednisolone, cyclophosphamide and plasmapheresis were started. Patient could be extubated without complications shortly. No hemoptoe appeared and kidney function was preserved.

Discussion: 90% of patients with anti-GBM disease present with renal involvement; in 20-65% additional pulmonary involvement is seen. However, isolated pulmonary disease is rare and happens predominantly in people aged <30 years. Our patient presented without hemoptoe or signs of renal involvement, but due to a “non-responding pneumonia work-up” anti-GBM was detected and patient could be treated accordingly.

Take Home Message: in case of non-responding pneumonia think of auto-immune disorders, anti-GBM included, even in older patients with absence of hemoptoe or renal involvement.

Literature:

1. Povoa P. et al. How to approach a patient hospitalized for pneumonia who is not responding to treatment? Intensive Care Med. 2025 Aug;51(8):1570-1571.
2. Polman J. et al. An uncommon presentation of a rare disease: a case of anti-glomerular basement membrane disease without renal involvement. Respir Med Case Rep. 2020 Nov 7;31:101282.

EP06

Breaking the viscous cycle: Rapid triglyceride reduction using plasmapheresis in severe hypertriglyceridemia induced necrotizing pancreatitis

1. van Wijngaarden1, J. Borst1, E. K. Haspels1

1 Department of Intensive Care, Martini Hospital Groningen, Groningen, the Netherlands

Teaser Case report

Een 42-jarige man presenteerde zich met een necrotiserende pancreatitis en een melkachtige laag in het bloed welke een ernstige hypertriglyceridemie verraadde. Een enkele sessie plasmaferese zorgde voor significante daling van serumtriglyceriden en, gecombineerd met ondersteunende therapie, tot klinische en biochemische stabilisatie. Dit benadrukt de relevantie van deze weinig gebruikte behandeloptie bij een vorm van pancreatitis met een verhoogd complicatierisico.

Case report

Introduction

Hypertriglyceridemia (HTG) is the third major cause of acute pancreatitis (AP). HTG ranges from mild to severe and can lead to HTG-induced pancreatitis which tends to be more severe than pancreatitis of other causes. There are higher rates of complications, ICU admissions, and recurrent or chronic pancreatitis – especially when triglycerides exceed 33.9 mmol/L (1). Treatment focuses on supportive care and rapidly lowering triglycerides by addressing underlying causes and implementing insulin-therapy and/or plasmapheresis (1).

Case Presentation & Diagnostic Assessment

A 42-year-old male, without priors , was presented at the Emergency Department with abdominal pain, vomiting, and constipation persisting for four days. Upon arrival, he was tachycardic, hypotensive, tachypneic and developed a fever (table 1).

Although largely disqualified due to extreme hypertriglyceridemia, initial laboratory testing was suggestive of de novo diabetes mellitus with diabetic ketoacidosis (DKA) (Table 1). Fluid resuscitation and insulin therapy were initiated, after which the patient was transferred to the ICU. Additional computed tomography showed early-stage necrotising pancreatitis, which was considered to be secondary to hypertriglyceridemia.

Treatment and Patient Outcome

Plasmapheresis was initiated alongside supportive care to prevent progressive organ dysfunction and complications such as hyperviscosity syndrome. One round of plasmapheresis resulted in a reduction of serum triglycerides from 67.7 to 8.07 mmol/L (Figure 1), after which the patient stabilised both clinically and biochemically in the following four days. After the ICU, the patient remained hospitalised for eight more days, and was discharged as an insulin-dependent diabetic.

Discussion

In HTG pancreatitis, breakdown of triglycerides releases toxic free fatty acids that cause acinar injury, inflammation and microcirculatory pancreatic ischemia (1). Although clinical trials are lacking in critically ill populations, extracorporeal elimination of large lipoproteins can reduce triglyceride levels by 50-97% in a single procedure, like in our case, and may prevent further organ damage in patients with signs of significant organ dysfunction, progressive systemic inflammation or lactic acidosis (1-2).

Furthermore, insulin therapy is believed to reduce triglyceride levels by activating lipoprotein lipase. Though initiated to treat DKA in our case, insulin may have been beneficial for hypertriglyceridemia as well (1).

Conclusion

Besides supportive care and fluid resuscitation, plasmapheresis rapidly reduces triglyceride levels and may prevent further organ damage in patients with severe HTG pancreatitis and adverse signs.

References

•  Yang AL, McNabb-Baltar . (2020). Hypertriglyceridemia and acute pancreatitis. Pancreatology, 20(5), 795-800.

Padmanabhan A, et al.. (2019). Guidelines on the Use of Therapeutic Apheresis in Clinical Practice - Evidence-Based Approach from the Writing Committee of the American Society for Apheresis: The Eighth Special Issue.. J Clin Apher, 34(3), 171-354.

EP07

Manifestation of mitral valve insufficiency in a young woman during diving: more than just a case report of immersion pulmonary edema

E.J. Ebbers1, D. Ramnarain1, B.M. Szabó1

1 ElisabethTweesteden Hospital, Tilburg, the Netherlands

Teaser Case report

Het begon als een ontspannen scuba duik in koud water en eindigde met een falend hart. In deze unieke case tonen we aan de hand van echobeelden hoe een gezonde duikster levensbedreigend immersion pulmonary edema (IPE) ontwikkelde, met mitralisklepinsufficientie en myocarddysfunctie, die volledig herstelden na snelle ondersteuning. Tijdige herkenning van IPE en inzicht in voorbijgaande cardiale veranderingen kunnen levens redden.

Case report

Introduction

Swimming and diving in cold open water can cause immersion pulmonary edema (IPE), a type of non-cardiogenic pulmonary edema, which is a life threatening condition if left untreated and can cause death if symptoms are not recognized (1). IPE is characterized by symptoms like dyspnea, hypoxia, crackles, and hemoptysis, which usually begin during the dive. It is thought to result from elevated pulmonary capillary pressure. Water compression and cold-induced venoconstriction shift blood centrally, increasing preload and capillary pressure. Combined with negative airway pressure during breathing, these factors promote fluid transudation into the alveoli.

Case

We present a well-documented case of IPE after scuba diving with multiple high-quality echocardiographic images portraying the evolution and resolution of mitral valve regurgitation. A 45-year old female experienced dyspnoea and respiratory distress after scuba diving for 20 minutes in cold water at 5 meter depth. She presented to the emergency department with clinical and radiological signs of pulmonary edema and she was hypotensive and hypoxic. Echocardiogram showed moderately reduced left ventricular function and moderate mitral valve regurgitation. After supportive therapy, i.e. supplemental oxygen, vasoactive medication and low dose diuretics, she fully recovered and only trivial mitral valve regurgitation persisted. Repeated echocardiograms depict this gradual resolution of mitral valve regurgitation (figures 2-4).

Discussion

IPE typically occurs in healthy athletes, but some cases involve individuals with pre-existing cardiopulmonary conditions. Risk factors include hypertension, overhydration before exercise, female sex, age over 50 years, cold water exposure and pre-existing cardiac conditions, such as a hypertrophic left ventricle (2). IPE tends to recur, suggesting predisposition in certain individuals (3). Subclinical or trivial mitral valve regurgitation may be a novel risk factor (4). Reversible myocardial dysfunction (RMD), when defined as coexistence of elevated cTnT levels with electrocardiographic and/or echocardiographic abnormalities, has been described before in the setting of IPE (5). Treatment of IPE usually consist of supportive oxygen, diuretics, sometimes also with β2 agonist. Non-invasive positive pressure ventilation can reduce left ventricular preload and accelerate clearance of hydrostatic pulmonary edema (6). NPPV was considered, but not deemed necessary in regards to our patient.

Conclusion

A routine cold-water immersion turned a trivial mitral valve regurgitation into a tipping point, quickly resulting in immersion pulmonary edema. This case reminds us subtle cardiac vulnerabilities matter and rapid recognition can save lifes, but it also triggers the question: how can we better predict which divers are at risk of surfacing in distress?

References

•  Kumar M, Thompson PD. A literature review of immersion pulmonary edema. Phys Sportsmed [Internet]. 2019;47(2):148–51. Available from: http://dx.doi.org/10.1080/00913847.2018.1546104
• Wilmshurst P. Immersion pulmonary edema. Chest [Internet]. 2021;159(5):1711–2. Available from: http://dx.doi.org/10.1016/j.chest.2020.12.017
• Peacher DF, Martina SD, Otteni CE, Wester TE, Potter JF, Moon RE. Immersion pulmonary edema and comorbidities: case series and updated review: Case series and updated review. Med Sci Sports Exerc [Internet]. 2015;47(6):1128–34. Available from: http://dx.doi.org/10.1249/MSS.0000000000000524
• Beck TP, Tsipis N, Kisslo JA, Rivera JD, Armour AC, Moon RE. Immersion-induced mitral regurgitation: A novel risk factor for swimming-induced pulmonary edema. Chest [Internet]. 2022;161(3):e137–43. Available from: http://dx.doi.org/10.1016/j.chest.2021.10.018
• Gempp E, Louge P, Henckes A, Demaistre S, Heno P, Blatteau J-E. Reversible myocardial dysfunction and clinical outcome in scuba divers with immersion pulmonary edema. Am J Cardiol [Internet]. 2013;111(11):1655–9. Available from: http://dx.doi.org/10.1016/j.amjcard.2013.01.339
• Seiler C, Kristiansson L, Klingberg C, Sundh J, Braman Eriksson A, Lundeqvist D, et al. Swimming-induced pulmonary edema: Evaluation of prehospital treatment with CPAP or positive expiratory pressure device. Chest [Internet]. 2022;162(2):410–20. Available from: http://dx.doi.org/10.1016/j.chest.2022.02.054

EP08

A patient with computed tomography (CT) findings consistent with subarachnoid hemorrhage without evidence of hemorrhage

Marthe Sliepenbeek1, Issam Boukrab1, Angela Kotsopoulos1

1 Department of Intensive Care, Elisabeth Tweesteden Hospital, Tilburg, the Netherlands

Teaser Case report

Een 57-jarige vrouw presenteerde zich met een fulminante bacteriële meningitis. Op de CT-scan van het cerebrum werd een beeld van een subarachnoïdale bloeding gezien welke gedurende opname bleek te berusten op een pseudo subarachnoïdale bloeding. Deze casus illustreert hoe een acute bacteriële meningitis kan leiden tot een vals-positieve diagnose voor een subarachnoïdale bloeding met risico op onjuiste behandeling

Case report

Introduction

A pseudo subarachnoid hemorrhage (pSAH) is a phenomenon in which characteristic findings of SAH on brain CT-scan are seen without evidence of hemorrhage on MRI, autopsy or cerebrospinal fluid analysis. (1-2) In this case, acute bacterial meningitis caused the radiographic mimic of SAH on the CT scan.

Case presentation

A 57-year old woman presented with decreased consciousness. The day before admission she complained about left sided ear pain and headache. Physical exam showed neck stiffness and left sided otorrhea. She had a septic shock, disseminated intravascular coagulation (DIC) and multi-organ failure. A brain CT-scan showed fading of the left middle ear and minimal SAH left occipital without vascular pathology on CT-angiography. The patient deteriorated fast and intubation was performed. An acute bacterial meningitis was suspected. A lumbar puncture for analysis of cerebrospinal fluid was too risky due to DIC. She was administered dexamethasone, ceftriaxone and acyclovir.

Soon after admission the brainstem reflexes became absent. A follow-up CT-scan showed cerebral edema and significant progression of the SAH with diffuse, symmetric hyperdensity involving the basal cisterns, lateral fissures, and cortical sulci, consistent for a pSAH considering the clinical history (Figure 1). Blood- and otorrhea cultures were positive for Haemophilus Influenza.

During admission the comatose state persisted and multi-organ failure aggravated. Considering the poor prognosis, the treatment was discontinued after which the patient died.

Figure 1: CT-scan on first day of admission at the ICU. Discussion

Different mechanisms result in the pSAH pattern in this patient. One explanation is leakage of

protein-rich exudate into the subarachnoid space due to breakdown of the blood-brain-barrier by toxins and inflammatory cells in the cerebrospinal fluid. (1, 3, 4) Another explanation is contrast

extravasation into the subarachnoid space after CT-angiography due to this impaired blood-brainbarrier. (1,2)

This case highlights the importance of correct interpretation of CT-imaging in correlation to the clinical findings, avoiding false-positive SAH in patients and therefore incorrect treatment.

References

•  Morgan E. Fretwell, Naresh Mullaguri, Sanjeev Sivakumar, Mike Knipfing. (2022). Pseudo Subarachnoid Hemorrhage Sign in Bacterial Meningitis in a Patient Presenting With Acute Ischemic Stroke: A Novel Radiological Clue to Rapid Diagnosis. Cureus , 14(5).
• Chun-Yu Lin, Ping-Hong Lai, Jui-Hsun Fu, Po-Ching Wang. (2013). Pseudo-Subarachnoid Hemorrhage: A Potential Imaging Canadian Association of Radiologists Journal, 65(3).
• Jozsef Lorant Lang, Paul L. Leach, John A. Emelifeonwu, Safquat Bukhari. (2013). Meningitis presenting as spontaneous subarachnoid haemorrhage (pseudo-subarachnoid heamorrhage). European Journal of Emergency Medicine, 20(2).

EP09

Sudden blueness a harbinger of a silent killer

M.Verrips1, A.M.M. Kotsopoulos1, J.H. van Oers1

1 Department of Intensive Care, Elisabeth TweeSteden Hospital, Tilburg, the Netherlands

Teaser Case report

Capnocytophaga canimorsus bacteriën, overgedragen via honden of katten, kunnen onverwacht ernstige infectie veroorzaken. Twee beschreven casussen bij immuun competente volwassen tonen een fulminant beloop met multiorgaanfalen en perifere cyanose. Snelle herkenning en directe antibiotische behandeling is cruciaal voor de overleving.

Case report

Introduction:

Capnocytophaga canimorsus is a gram-negative rod and a commensal in the oral flora of dogs and cats. In humans it can cause severe infections, including fulminant sepsis in immunocompetent patients as illustrated by the two cases reported (1).

Case presentation and clinical findings:

Case 1, a 42-year-old man presented with acute onset general illness, gastrointestinal symptoms and painful peripheral cyanosis (Image 1 and 2). He developed symptoms the day before presentation. Additional blood tests showed thrombocytopenia, leukocytosis with neutrophil predominance, and disseminated intravascular coagulation (DIC). Empirical treatment with antibiotics (ceftriaxon and tobramycin) was started immediately. One day later, blood cultures confirmed C. canimorsus. Only after multiple repetitive questioning, the patient recalled a possible minor dog bite three days before admission. The clinical status improved, and the patient was transferred to the general ward.

Case 2, a 64-year-old woman presented at the ER with somnolence, extreme facial cyanosis and septic shock with multiorgan failure. A third-party interview indicated gastrointestinal problems and general illness for two days. Laboratory tests showed metabolic acidosis, thrombocytopenia, leukocytosis and DIC. The patient had dogs, but no confirmed history of a bite. Despite aggressive supportive care, including mechanical ventilation and broad spectrum antibiotics (ceftriaxon and tobramycin), the patients’ condition deteriorated rapidly. On day of admission, blood smears revealed gram-negative rods enclosed in leucocytes, typically seen in C. canimorsus. Despite treatable diagnosis, the patient developed refractory septic shock and died within twenty-four hours after admission.

Discussion: These cases highlight the fulminant nature of C. canimorsus infection, which can occur even in immunocompetent individuals (1). In both cases, severe painful peripheral cyanosis was observed and C. canimorsus was identified as the cause of rapid-onset sepsis only after several days. None of the patients had initially evidence of a dog bite or animal exposure at presentation, delaying the recognition of this infection. Therefore, C. canimorsus should be considered in patients with gastrointestinal symptoms, rapidly progressive sepsis especially when accompanied by peripheral or facial cyanosis. Prompt recognition and initiation of appropriate antibiotic, mainly beta-lactam inhibitors, advance cephalosporins or carbapenems, therapy are critical in improving patient outcomes.

References

1.  Mally, M., Shin, H., Paroz, C., Landmann, R., & Cornelis, G. R. (2008). Capnocytophaga canimorsus: a human pathogen feeding at the surface of epithelial cells and phagocytes. PLoS pathogens, 4(9), e1000164

EP10

Levamisole-adulterated cocaine, a deathly outcome

I.B. van Ingen1, P. van Vliet1

1 Department of Intensive Care, Haaglanden Medisch Centrum, The Hague, the Netherlands

Teaser Case report

Door de toenemende prevalentie van levamisol-verontreinigde cocaïne worden steeds vaker ernstige complicaties gerapporteerd. Dit artikel beschrijft een fatale casus van diepe pancytopenie, gecompliceerd door een pulmonale sepsis. Haaranalyse bevestigde uiteindelijk een diagnose per exclusie: levamisol-geïnduceerde aplastische anemie. De bevindingen onderstrepen de diagnostische complexiteit en de noodzaak van verhoogde klinische alertheid voor deze potentieel irreversibele en fataal verlopende toxiciteit.

Case report

With the rising prevalence of levamisole-adulterated cocaine, serious complications are increasingly encountered (1). We present a fatal case of profound and possibly irreversible pancytopenia attributed to levamisole-adulterated cocaine, highlighting the diagnostic difficulties and the need for heightened clinical awareness.

A 40-year-old patient with a history of drug abuse, episodic psychosis, and anemia of unclear origin presented to the emergency department with chest pain. Unexpectedly, he was found to have profound pancytopenia, including a hemoglobin level of 2.2 mmol/L, a platelet count <5 × 10 /L, and a leukocyte count of 3.0 × 10 /L. Because of severe agitation, he required sedation, endotracheal intubation, and admission to the intensive care unit.

During admission, inflammatory markers increased and he developed sepsis with a CT-confirmed pulmonary focus. Despite repeated red blood cell and platelet transfusions, empirical broad-spectrum antibiotics, and filgrastim therapy, his clinical condition progressively deteriorated the following 14 days. Bone marrow biopsy revealed marked aplasia, and serial laboratory studies showed a persistent absence of hematopoiesis. Extensive diagnostic evaluation identified only a low quantity of Klebsiella species in bronchoalveolar lavage fluid, which was deemed insufficient to account for the clinical severity. Ultimately, the patient developed refractory septic shock and died despite maximal supportive treatment.

In this case, common causes of pancytopenia were excluded. Given the patient’s known drug abuse, exposure to levamisole-adulterated cocaine was suspected. Because levamisole has a short serum and urine half-life, the diagnosis was confirmed through the toxicological analysis of the patient’s hair. (2) Although definitive causality cannot be proven, the findings strongly suggest levamisole-induced aplastic anemia.

We hypothesize that the cause of death was overwhelming sepsis facilitated by profound immunodeficiency. No clinically plausible pathogens were identified, but opportunistic infection or reactivation remains likely. Currently, discontinuation of levamisole exposure and supportive care constitute the only available management strategies for levamisole-induced cytopenias. Thus, in this case, earlier identification would not have altered therapeutic decisions.

Levamisole-contaminated cocaine has become a growing public health concern and is known to cause severe vasculitis, neutropenia and agranulocytosis. (1) In this patient, pancytopenia occurred without the characteristic vasculitis. The lack of hematopoietic recovery despite abstinence suggests significant and possibly irreversible toxicity. These findings emphasize the need for sustained clinical vigilance, even in the absence of vasculitic manifestations.

• Quick scan rapportage levamisol 2014 update https://www.rivm.nl/sites/default/files/2018-11/CAM%20Quick%20Scan%20rapportage%20levamisol%20update%20finale%20versie.pdf
• Lazareth H, Peytavin G, Polivka L, Dupin The hairy-print for levamisole-induced vasculitis. BMJ Case Rep. 2012 Aug

References

• Coördinatiepunt Assessment en Monitoring nieuwe drugs (CAM). (2015). Quick scan rapportage levamisol 2014 update. https://www.rivm.nl/sites/default/files/2018-

11/CAM%20Quick%20Scan%20rapportage%20levamisol%20update%20finale%20versie.pdf

• Lazareth H, Peytavin G, Polivka L, Dupin . (2012). The hairy-print for levamisole-induced vasculitis.. BMJ Case Reports.

EP11

Navigating intermittent hemodialysis in heart failure: proof of concept for LVAD patients

V.E.M. Grupa1, F.S. van den Brink1, W.M. Michels2, C.V. Elzo Kraemer1

1 Department of Intensive Care, Leiden University Medical Centre, Leiden, the Netherlands

2 Department of Nefrology, Leiden University Medical Centre, Leiden, the Netherlands

Teaser Case report

Hemodynamic monitoring and management of instability during intermittent hemodialysis (IHD) outside the ICU can be particularly challenging in patients supported by a left ventricular assist device (LVAD). It complicates noninvasive monitoring of vital parameters and quick withdrawal of large fluid volumes. This case demonstrates proof of concept for IHD in an LVAD patient, with and without hemodynamic monitoring.

Case report

A 30-year-old patient was admitted to the ICU requiring renal replacement therapy (RRT). His medical history included cardiogenic shock with idiopathic heart failure, ultimately necessitating LVAD implantation, followed by persistent renal failure attributed to a cardiorenal syndrome with a component of focal segmental glomerulosclerosis (FSGS). Post-LVAD implantation and after a brief period of expectant management, the patient ultimately required renal replacement therapy. In this case, peritoneal dialysis (PD) had been selected as dialysis modality.

Shortly before this admission, the patient had exhibited a recurrent PD peritonitis and it was decided that the PD catheter had to be extracted. Although the patient remained clinically stable, the anticipated three-week interval before placement of a PD catheter and the catheter being ready for clinical use required bridging therapy with continuous RRT in the ICU, as the patient had no residual renal function. During this ICU admission, the patient remained hemodynamically stable and requested greater mobility. Therefore, though experience with IHD in LVAD supported patients is limited in both clinical practice and literature(1), a trial of IHD was undertaken.

The first IHD session was performed in the ICU with continuous invasive arterial blood pressure monitoring, alongside close clinical assessment. As shown in the accompanying figure, mean arterial pressure and other vital parameters remained stable throughout the four hour session, during which approximately 2 liters of fluid were removed. The patient experienced no symptoms of hypoperfusion and no adjustments to LVAD settings were required. A second IHD session, performed several days later without invasive monitoring and relying only on clinical observation and LVAD parameters (including the pulsatility index) reflecting the conditions expected on the IHD-unit. This session also proceeded uneventfully and the patient was subsequently transferred back to the ward for IHD awaiting resumption of PD.

This case demonstrates that IHD can be performed safely in a carefully selected, hemodynamically stable patient with LVAD support, it highlights the potential role of IHD as a feasible bridging strategy in patients unsuitable for immediate PD and may support broader consideration of this approach in similar clinical scenarios.

1. daSilva-deAbreu A, Faaborg-Andersen C, Joury A, Tutor A, Desai S, Eiswirth C, et al. Outcomes of Patients With Left Ventricular Assist Devices Requiring Intermittent Hemodialysis: Single-Center Cohort, Systematic Review, and Individual-Participant Data Meta-Analysis. Current Problems in Cardiology. 2024 Jan;49(1).

References

daSilva-deAbreu A, Faaborg-Andersen C, Joury A, Tutor A, Desai S, Eiswirth C, et al.. (2024). Outcomes of Patients With Left Ventricular Assist Devices Requiring Intermittent Hemodialysis: Single-Center Cohort, Systematic Review, and Individual-Participant Data Meta-Analysis. Current Problems in Cardiology, 49(1).

EP12

A non-fatal case of monomicrobial necrotizing soft tissue infection caused by E. coli in an immunocompromised patient with IgA – nephropathy and kidney transplantation

Amber Maliepaard1, Erik Bathoorn2, Joost Hoekstra3, Janesh Pillay1, Charlotte van den Berg1

• Department of Intensive Care, University Medical Center Groningen, Groningen, the Netherlands
• Department of Medical Microbiology and Infection Prevention University Medical Center Groningen, Groningen, the Netherlands
• Department of Trauma surgery, University Medical Center Groningen, Groningen, the Netherlands

Teaser Case report

Necrotiserende weke delen infecties (NWDI) zijn levensbedreigend en vereisen snelle interventie. Wij beschrijven een zeldzame type III NWDI door Escherichia coli bij een immuungecompromitteerde niertransplantatiepatiënt, behandeld met complementfactor B-remming. Vroege herkenning, onmiddellijke stopzetting van immunosuppressie en agressieve chirurgie resulteerden in overleving. Dit benadrukt het belang van alertheid op Gram-negatieve NWDI bij transplantatiepatiënten.

Case report

Background

Necrotizing soft tissue infections (NSTIs) are rapidly progressive infections with high mortality. Type III NSTIs, caused by Gram-negative bacteria such as Escherichia coli, are rare but associated with mortality rates up to 100%, particularly in immunocompromised patients. Complement inhibition may further impair host defense against invasive Gram-negative pathogens.

Methods

We report a case of monomicrobial E. coli NSTI in a 43-year-old woman with IgA nephropathy and kidney transplantation, treated with iptacopan (a complement factor B inhibitor) and intensified immunosuppression. Clinical course, microbiological findings, and therapeutic interventions were analyzed. A literature review identified previously reported cases of E. coli NSTI in kidney transplant recipients.

Results

Nine days after admission, the patient developed rapidly expanding perianal erythema and septic shock. Initial surgical exploration showed no necrosis; however, Gram staining revealed Gram-negative bacteria. Within hours, extensive necrosis developed, requiring repeated debridements, diverting colostomy, and vacuum-assisted closure therapy. Aggressive resuscitation, broad-spectrum antibiotics, and intravenous immunoglobulins were administered. Immunosuppressive therapy, including iptacopan, was discontinued immediately. Cultures identified E. coli ST69 (O15:H18), a uropathogenic lineage without specific necrosis-associated toxins. Despite severe shock, the patient survived and was transferred to the ward. After >30 days she is alive and well. Literature review revealed five other cases of E. coli NSTI in kidney transplant patients, with 40% survival; all infections occurred in the lower extremity.

Conclusion

This case illustrates that early recognition, immediate cessation of complement inhibition and immunosuppression, and aggressive surgical and supportive therapy can be lifesaving in type III NSTI. Clinicians should maintain high suspicion for Gram-negative NSTI in transplant recipients, including those receiving complement inhibitors.

EP13

A fortuitous Twist: achalasia and the hidden threat of Mycobacterium fortuitum

S.M. Bindraban1, L. Hughes2, C.H.S.B. van den Berg1, T.C. Veenstra1, D. Bathoorn2, B. Sinha2

• Department of Intensive Care, University Medical Center Groningen, Groningen, the Netherlands
• Department of Microbiology, University Medical Center Groningen, Groningen, the Netherlands

Teaser Case report

Langdurige IC-opname vanwege een atypische pneumonie met een niet gediagnosticeerde achalasie, leidde tot een zeldzame diagnose: een infectie met een Mycobacterium houstonense. De Niet-Tuberculeuze Mycobacterie werd geïdentificeerd in bloedkweken en broncho-alveolaire lavage, waarna gerichte antibiotica en endoscopische behandeling tot volledig herstel leidden. Deze casus onderstreept hoe cruciaal het is om NTM-diagnostiek te overwegen bij oesofageale motiliteitsstoornissen en atypische pneumoniae.

Case report

This case concerns a 44-year-old Polish woman with a history of chronic swallowing difficulties who presented with bilateral pneumonia (PSI Class III), persistent fever, general malaise, hemodynamic instability, and fluctuating elevated CRP levels despite empirical antibiotic therapy. Further investigation revealed significant oesophageal achalasia of unknown aetiology.

During treatment, the patient experienced a hypoxic cardiac arrest, necessitating prolonged intensive care unit (ICU) management. Mycobacterium houstonense was detected in blood and bronchoalveolar lavage samples, determining the diagnosis of disseminated pneumonia. In the absence of other identifiable pathogens, treatment was changed to imipenem, amikacin, and levofloxacin based on susceptibility results. During the course of her ICU stay, corticosteroids were administered due to suspected organizing pneumonia as a concurrent complication.

Addressing the suspected underlying pathologic factor, achalasia, oral intake was prohibited, and the patient underwent oesophageal balloon dilation followed by Botox injection. After extended period of mechanical ventilation and ICU care, the patient was transferred to the pulmonology ward to continue treatment. With the continuation of her therapy, she made a full recovery.

Mycobacterium houstonense belongs to the Mycobacterium fortuitum group, an organism first identified in Houston, Texas.1 Although members of the M. fortuitum group rarely cause severe disease in humans, they can lead to opportunistic infections such as skin, pulmonary, and soft tissue infections in immunocompromised individuals.2 These rapidly growing mycobacteria are saprophytic organisms often isolated from soil, water, and certain animals, including fish.2

The association between achalasia and M. fortuitum group infections was first reported in 1953, although its exact mechanism remains unclear.4 Oesoghageal food stasis in achalasia creates a lipid-rich environment that may facilitate the transition of non-tuberculosis mycobacteria (NTM) from non-pathogenic to pathogenic. This combined with the impairment of oesophageal sphincter function and chronic aspiration may contribute to the development of severe pulmonary complications, such as pneumonia and bacteremia as was seen in this case. 3,4

This case highlights the importance of considering NTMs in patients with underlying oesophageal disorders and atypical pneumonias. This life-threatening disease entity should be treated with oesphageal source controle and adequate antibiotics.

References

•  Mycobacterium houstonense and its classification as a species in the Mycobacterium fortuitum complex. (2004). International Journal of Systematic and Evolutionary Microbiology, 54(5), 1653–1656. DOI: 10.1099/ijs.0.02779-0
• Sharma, S.K. (2020). Epidemiology, diagnosis & treatment of non-tuberculous mycobacterial disease. Indian J Med Res, 152, 185–226. DOI: 4103/ijmr.IJMR_902_20
• Varghese, G., et al. (1988). Fatal Infection with Mycobacterium Fortuitum associated with oesophageal achalasia. Thorax, 43, 151–152
• Church, C., et al. (2006). Non-tuberculous mycobacteria masquerading as aspiration pneumonia in patients with gastrointestinal problems. Resp Med, 100, 1663–1665

EP14

Coronary Arterial Thrombosis as a Severe Complication of Hyperosmolar Hyperglycaemic State: A Case Requiring Multi-Level Mechanical Support

O.A. Welleman1, F.S. van den Brink1, J.J. Maas1, J.E. López Matta1, J.A. Janson1, M. Palmen3, J.M. Montero-Cabezas2, C. Elzo Kraemer1

• Department of Intensive Care, Leiden University Center, Leiden, the Netherlands
• Department of Cardiology, Leiden University Center, Leiden, the Netherlands
• Department of Thoracic, Leiden University Center, Leiden, the Netherlands

Teaser Case report

Een patiënt met HHS ontwikkelde ernstige arteriële coronaire trombose en diepe cardiogene shock. We beschreven het verloop en de behandeling met geavanceerde mechanische ondersteuning met Impella, V-A ECMO en LVAD. De casus onderstreept het belang van vroege herkenning van trombotische complicaties bij HHS en tijdige inzet van mechanische ondersteuning.

Case report

A 30-year-old man with no relevant medical history was admitted to a referring hospital with a hyperosmolar hyperglycaemic state (HHS) with initial glucose 93 mmol/L and serum osmolality 388 mOsm/kg. Work-up revealed previously undiagnosed latent autoimmune diabetes in adults (LADA) confirmed with positive GAD autoantibodies. He reported progressive unexplained weight loss over the preceding eight months, which he had compensated with excessive intake of sugar-rich foods.

The admission ECG showed ST-segment elevations consistent with an anterior wall myocardial infarction, although the patient did not experience chest pain. Transthoracic echocardiography demonstrated anterior wall akinesia, prompting urgent coronary angiography. This revealed a thrombotic occlusion of the LAD and its anterolateral branch, with successful balloon dilation of the latter but persistent LAD occlusion. During the procedure, the patient developed profound cardiogenic shock with bradycardia and hypotension, requiring atropine, adrenaline boluses and initiation of norepinephrine. He was intubated due to respiratory deterioration. Given refractory cardiogenic shock (SCAI stage D) with concomitant respiratory and renal failure, he was transferred to a referral center.

On arrival, an Impella CP was implanted, followed by thrombectomy of the LAD, yielding substantial thrombus burden but without flow restoration. Echocardiography showed reduced left ventricular function with preserved right ventricular function. Recurrent haemodynamic instability necessitated escalation to femoral-femoral V-A ECMO, later converted to subclavian cannulation due to hyperperfusion syndrome of the cannulated limb with early compartment syndrome. Over time, he stabilised, underwent gradual decongestion with Continuous Veno-Venous Hemofiltration (CVVH), and showed good neurological recovery.

A staged ECMO weaning trial under maximal Impella support was successful. However, due to persistent LV dysfunction and renal failure, a left ventricular assist device (LVAD) was implanted as bridge-to-decision, and the patient was successfully extubated three weeks after transfer. Considering technical challenges regarding vascular access and the risk of failure upon decannulation, V-A ECMO was maintained as a bridge to the LVAD implantation. After full renal recovery, the patient successfully underwent heart transplantation three years later.

Extensive diagnostic evaluation including antiphospholipid syndrome serology, whole-body CT imaging and thrombophilia screening revealed no explanation for the high thrombogenic status. The thrombotic burden was ultimately attributed to profound hyperosmolarity in the context of HHS. This case illustrates how rapidly progressive cardiogenic shock in the context of HHS may require swift escalation through multiple layers of mechanical circulatory support, and reinforces the need for heightened awareness for arterial thrombotic complications in HHS.

EP15

From diabetic ketoacidosis to catastrophic antiphospholipid syndrome; an unexpected clinical evolution

C.B. Nijboer1, P.N. Günkel1, M.J.M.M van der Steen-Dieperink1

1 Department of Intensive Care, Martini Hospital Groningen, Groningen, the Netherlands

 Teaser Case report

Een patiënte presenteert zich met een ernstige diabetische ketoacidose op de IC, waarna zij trombo-embolische complicaties met darmischemie en meerdere arteriële occlusies ontwikkelt. Bij de work-up van de verhoogde stollingsneiging blijkt patiënte een catastrofaal antifosfolipidensyndroom te hebben. Ondanks behandeling met plasmaferese, corticosteroïden en therapeutische antistolling, overlijdt patiënte. Vroegtijdige herkenning van catastrofaal antifosfolipidensyndroom kan helpen om tijdige interventie te starten (1).

Case report

A 79-year-old female with a history of type 1 diabetes mellitus, hypertension, and asthma presented to the hospital with a diabetic ketoacidosis with symptoms of tachypnea (30/min), hypotension (100/30 mmHg) and confusion. Her glucose levels were dysregulated because her monitor was malfunctioning. Her glucose level was 73 mmol/L and her arterial blood gas showed a metabolic acidosis with a pH of 7.02. She was transferred to the Intensive Care Unit (ICU) for intravenous insulin therapy and rehydration.

On the second day of admission, her condition deteriorated rapidly. She developed bloody stools, and her right hand became cold and pale. A Doppler ultrasound revealed a thrombus in the right brachial artery. A CT scan of the abdomen was performed, showing diffuse jejunal wall thickening and lack of enhancement, suggestive of ischemia. On the following day, another CT-scan of the brain, thorax and abdomen was performed, revealing extensive thrombosis of the superficial femoral artery and popliteal artery. Despite adequate resuscitation and management, her hypotension persisted.

Because of the thromboembolic complications, a work-up for hypercoagulability was done. Disseminated intravascular coagulation and heparin-induced thrombocytopenia were ruled out. A transesophageal echocardiogram showed no embolic source and a preserved cardiac function. Subsequent laboratory tests, showed an elevated lupus anticoagulant (LAC). The concomitant presence of >2 embolisms suggested the diagnosis of catastrophic antiphospholipid syndrome (CAPS).

15 cm of the jejunum were surgically resected and an emergency thrombectomy of the affected arteries was performed. After the diagnosis of CAPS, therapeutic nadroparin, methylprednisolone, and plasma exchange therapy were started. Despite aggressive treatment, the patient's condition continued to worsen. After discussions with her family, the decision was made to withdraw life support, after which she passed away.

CAPS is a diagnostic challenge, particularly in patients with other complicating factors such as DKA and pre-existing comorbidities

(2). In this patient, the onset of multiple ischemic events and the presence of antiphospholipid antibodies pointed towards CAPS as the underlying cause, although the precise trigger for the syndrome remains unclear. While improved diagnosis and treatment of CAPS have led to a reduction in mortality, it remains as high as 30% (3). In this case, aggressive treatment could only be initiated after systemic thromboembolic events had already presented. This underscores the need for early recognition of CAPS in patients with multiple organ ischemia, as timely management is essential in improving patient outcome (4).

References

•  Okunlola AO, Ajao TO, Sabi M, Kolawole OD, Eweka OA, Karim A, Adebayo TE.. (2024). Catastrophic Antiphospholipid Syndrome: A Review of Current Evidence and Future Management Practices. Cureus.
• Jacobs L, Wauters N, Lablad Y, Morelle J, Taghavi M. (2024). Diagnosis and Management of Catastrophic Antiphospholipid Syndrome and the Potential Impact of the 2023 ACR/EULAR Antiphospholipid Syndrome Classification Criteria. Antibodies (Basel).
• Rodríguez-Pintó I, Moitinho M, Santacreu I, Shoenfeld Y, Erkan D, Espinosa G, Cervera R; CAPS Registry Project Group (European Forum on Antiphospholipid Antibodies). (2016). Catastrophic antiphospholipid syndrome (CAPS): Descriptive analysis of 500 patients from the International CAPS Registry. Autoimmun Rev.
• Cervera R, Rodríguez-Pintó I, Colafrancesco S, Conti F, Valesini G, Rosário C, Agmon-Levin N, Shoenfeld Y, Ferrão C, Faria R, Vasconcelos C, Signorelli F, Espinosa G. (2014). 14th International Congress on Antiphospholipid Antibodies Task Force

Report on Catastrophic Antiphospholipid Syndrome. Autoimmun Rev.

EP16

Acute respiratory distress syndrome induced by paroxetine intoxication: A near fatal case

1. Beijnsberger1, T.C.C. Jaspers2, C.A. Boly1, J.A.H. van Oers1

• Department of Intensive Care, Elisabeth Tweesteden Hospital, Tilburg, the Netherlands
• Department of Pharmacy, Elisabeth Tweesteden Hospital, Tilburg, the Netherlands

Teaser Case report

Een paroxetine intoxicatie wordt gezien als veilig antidepressivum met milde symptomen bij een intoxicatie. Dit case report beschrijft een 70-jarige patiënte met een ernstige paroxetine intoxicatie, gecompliceerd door een acute respiratory distress syndrome (ARDS) en stijgende serumlevels van paroxetine gedurende het verblijf op IC. Het benadrukt de mogelijkheid van ernstig beloop en het belang van tijdige maag- en darmspoeling.

Case report

Selective serotonin reuptake inhibitors (SSRIs) are commonly prescribed antidepressants. Their pharmacological profile is considered relatively safe with fewer side effects and relatively mild symptoms in cases of intoxication compared to other antidepressants (1). This report describes a unique case of paroxetine intoxication resulting in acute respiratory distress syndrome (ARDS).

A 70-year old woman with a history of depression, alcoholism, and hypothyroidism was admitted to the emergency department after being found with altered consciousness. Empty medication containers were found at her bedside, including immediate-release paroxetine (30 mg). At presentation, her vital signs were stable, but neurological examination revealed a Glasgow Coma Scale (GCS) of E4M6V2, myoclonus, reduced attention span, and diminished tendon reflexes. Blood tests were unremarkable. An electrocardiogram revealed a prolonged QT interval of 647ms.

Given the clinical manifestations, a paroxetine intoxication was suspected. The patient was admitted to the intensive care unit (ICU) for close monitoring, where activated charcoal was administered. Despite initial supportive care, her respiratory status deteriorated, and ARDS developed. A chest radiograph revealed bilateral infiltrates, suggestive of ARDS. Intubation was required due to worsening hypoxemia. The patient developed hyperthermia and required norepinephrine support. A computerized tomography scan (CT) of the thorax and abdomen showed no signs of infection. Serotonin syndrome was suspected.

Paroxetine levels were measured in retrospect on day five. On the day of admission, the paroxetine levels were 4805 µg/L, well above lethal threshold (2). During admission, the paroxetine levels increased further, contrary to the expected pharmacokinetics, suggestive for an ongoing absorption (Figure 1). Bowel lavage was started due to possible pharmacobezoars. Further treatment focused on respiratory support and the management of ARDS with mechanical ventilation, prone positioning, and corticosteroids. After 21 days, the patient showed significant improvement. Her ARDS resolved, she was extubated and stable with nasal oxygen. Paroxetine levels eventually normalized, and she was discharged from the ICU after further rehabilitation. This case underscores that paroxetine overdose may present more severely than commonly documented in existing literature. Clinicians should remain vigilant for such presentations and initiate timely gastric and bowel lavage and close monitoring.

References

•  Bruggeman C, O'Day CS. Selective Serotonin Reuptake Inhibitor Toxicity. StatPearls. Treasure Island (FL)2025.
• Schulz M, Schmoldt A, Andresen-Streichert H, Iwersen-Bergmann S. Revisited: Therapeutic and toxic blood concentrations of more than 1100 drugs and other xenobiotics. Crit Care. 2020;24(1):195.

EP17

Vaak mild, nu fataal: snel progressieve infectieuze endocarditis door Granulicatella adiacens

N.D.M. Oude Avenhuis1, A. Gomes2, B. van Anrooij3, B.N.M. Sinha2, A. Oude Lansink - Hartgring1

• Department of Intensive Care, University Medical Center Groningen, Groningen, Nederland
• Department of Medical Microbiology and Infection Prevention, University Medical Center Groningen, Groningen, Nederland
• Department of Pathology and Medical Biology, University Medical Center Groningen, Groningen, Nederland

Teaser Case report

Een 20-jarige patiënte kan ’s ochtends plotseling geen enkele inspanning meer leveren. Door de ambulance wordt een vrouw in diepe shock aangetroffen. In het ziekenhuis wordt echografisch de waarschijnlijkheidsdiagnose infectieuze endocarditis (IE) gesteld. Patiënte overlijdt binnen enkele uren. De bloedkweken zijn snel positief voor Granulicatella adiacens. Een zeldzame verwekker die bekend staat om zijn lenta beloop.

Case report

A 20-year-old woman experienced general malaise for several days, progressing to severe dyspnea the morning of presentation. Primary assessment showed a patient in shock with kidney and liver failure, severe anemia and lactic acidosis. To differentiate types of shock, point-of-care ultrasound was performed, revealing a large vegetation on the mitral valve with valvular insufficiency. A contrast CT-scan showed no signs of bleeding nor other infectious foci. During in-hospital transport, the patient developed asystole for which advanced life support was performed. After return of circulation, suspicion of IE was confirmed by echocardiography (Figure 1). Given the severity of shock, Staphylococcus aureus was suspected as causing micro-organism and high-dose flucloxacillin was started. After ICU admission, circulatory support with vasopressors and renal replacement therapy were given to stabilize the patient for further treatment. Despite, she developed bradycardia with loss of circulation and died. Later, autopsy revealed severe destruction of the mitral valve (Figure 2).

The next day, blood cultures showed Granulicatella adiacens with a strikingly short time to detection (6-8 hours). Granulicatella (GRA) species, together with Abiotrophia (ABI), are fastidious gram-positive cocci which are part of the normal oral, gastrointestinal and genitourinary flora. They are facultative pathogens that can cause infections in immunocompetent and immunocompromised patients.1 GRA/ABI-IE represent around 1-3% of IE and the largest prospective multicenter cohort study reports a typical subacute presentation with better prognosis than viridans group streptococci-IE. Notably, GRA/ABI-IE is more common in younger patients.2

Detection of Granulicatella species can be difficult because of slow growth and specific nutritional requirements. The very fast time to positivity of this patient’s blood cultures, together with the peracute clinical presentation, raised questions whether this isolate had an abnormal virulence phenotype.

A very likely explanation for this atypical presentation of GRA-IE, is late clinical presentation of a young patient that compensated cardiac failure and shock for a prolonged period. This could also explain the exceptionally fast time to positivity indicative of a high bacterial load in her blood. The long prodromal phase was later confirmed by the patient’s mother, who indicated that she experienced fatigue for several weeks, a commonly reported symptom in subacute IE.

This case clearly demonstrates that a multidisciplinary approach is important to accurately diagnose and treat IE, especially when its presentation is atypical.

References

• Bokhari SFH, Bakht D, Amir M, Haris HM, Ain NU, Qureshi MS, Yousaf F, Yousaf R, Ali K, Javed MA, Awais MN, Zahid M, Shaukat M, Usman S, Hassan A, Ejaz M.. (2025). Granulicatella infections: Comprehensive review of an elusive opportunistic pathogen. World Journal of Clinical Cases.
• Téllez A, Ambrosioni J, Hernández-Meneses M, Llopis J, Ripa M, Chambers ST, Holland D, Almela M, Fernández-Hidalgo N, Almirante B, Bouza E, Strahilevitz J, Hannan MM, Harkness J, Kanafani ZA, Lalani T, Lang S, Raymond N, Read K, Vinogradova T, Woods CW, Wray D, Moreno A, Chu VH, Miro JM; International Collaboration on Endocarditis (ICE) . (2022). Clinical characteristics and outcome of infective endocarditis due to Abiotrophia and Granulicatella

compared to Viridans group streptococci. Journal of Infection.

EP18

Toxic epidermal necrolysis in a severely immunocompromised patient: a complex therapeutic dilemma

1. Meeder1, D. P. Boer1

1 Department of Intensive Care, Maasstad Hospital, Rotterdam, the Netherlands

Teaser Case report

Wij presenteren een casus van een ernstig immuungecompromitteerde patiënt die een fulminante toxische epidermale necrolyse (TEN) ontwikkelt tijdens behandeling voor tuberculose en HIV. We beschrijven het klinisch beloop, complicaties en multidisciplinaire interventies. De combinatie van TEN, opportunistische infecties en medicatiedilemma’s leidde tot aanzienlijke morbiditeit. Deze casus benadrukt de uitdagingen van therapiekeuzes bij ernstige immuunsuppressie.

Case report

Presenting symptoms

A 45-year-old male with recently diagnosed HIV infection (CD4 10*109/L) and isoniazid-resistant miliary tuberculosis was transferred to the burn unit of our hospital due to progressive skin lesions. In the preceding weeks, he had received multiple antituberculous agents, antiretroviral therapy, and other antimicrobials for different infections. The anti-tuberculous drugs were already stopped before transfer to our hospital. Over the next week skin lesions were progressive, with hypovolemia due to fluid loss and fever.

Clinical course and interventions on the intensive care unit (ICU)

The patient was transferred to the ICU of our burn unit for intensive monitoring, resuscitation, and temperature management. Within the first week, he developed 100% epidermal detachment with extensive mucosal involvement, requiring intubation and aggressive fluid therapy. Due to ongoing progression, antiretroviral therapy was discontinued because of suspected drug-induced TEN. His course was complicated by hypovolemic shock, acute kidney injury, and recurrent bloodstream infections, consistent with complete loss of skin barrier function. After stopping antituberculous and antiretroviral drugs, there was no evidence of active tuberculosis, though HIV viral load increased rapidly. No additional antiretroviral therapy was initiated during the ICU stay.

Over subsequent weeks, gradual re-epithelialization occurred, with residual defects in the axillae and gluteal region. The patient required prolonged sedation and tracheostomy. On day 40 the patient was transferred to a tuberculosis expertise center for restart of HIV and tuberculosis treatment with alternative regimens. At transfer, the patient was weaned from mechanical ventilation and free of active bacteremia.

Diagnosis

The diagnosis of TEN was confirmed by skin biopsy. TEN is an extremely rare life-threatening, medication-related condition requiring immediate withdrawal of the offending drug and intensive supportive care. Patients with advanced HIV infection and disseminated tuberculosis are particularly vulnerable, as treatment choices are complicated by overlapping toxicities, resistance, and (opportunistic) infections. This case illustrates the major therapeutic challenges of managing TEN in a profoundly immunosuppressed patient with drug-resistant tuberculosis, emphasizing the need for early recognition, multidisciplinary management, and careful balancing of essential anti-infective therapy against the risk of severe adverse drug reactions.

References

•  Joy Justice 1, Eric Mukherjee 2, Michelle Martin-Pozo 3, Elizabeth Phillips 4. (n.d.). Updates in the pathogenesis of SJS/TEN. Allergology International | Journal.
• Shah H, Parisi R, Mukherjee E, Phillips EJ, Dodiuk-Gad (n.d.). Update on Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: Diagnosis and Management. American Journal of Clinical Dermatology.

EP19

Underpressure? Under Pressure!

1. Roest1, H.F.E.M. Willems1, W. van der Tempel1

1 Department of Intensive Care, Ikazia Hospital, Rotterdam, the Netherlands

Teaser Case report

Acuut longoedeem kan cardiogeen of niet-cardiogeen zijn, maar bijzondere combinaties bestaan. Een respiratoir insufficiënte patiënte met overvullingsbeeld reageert slecht op Bi-level Positive Airway Pressure (BiPAP), waarna intubatie volgt. Laryngoscopie toont een supraglottische tumor, echocardiogram toont beeld passend bij een Tako Tsubo Cardiomyopathie (TTC). De combinatie negatieve-druklongoedeem met TTC benadrukt het belang van aanvullende diagnostiek en vereist een tweesporen therapeutisch beleid.

Case report

Introduction: Acute pulmonary edema can have a cardiac or non-cardiac origin, but mixed origins should be considered. We present a patient suffering of both negative pressure pulmonary edema (NPPE) and Takotsubo cardiomyopathy (TTC) induced edema.

Presentation: A 72-year-old woman, no known cardiac history, presented with acute dyspnoea. Patient did not use medication, was a smoker (50 pack years), and no provoking factors. Before admission supplemental oxygen, nebulization therapy, and hydrocortisone were administered.

Clinical findings: The patient was in respiratory distress, with inspiratory stridor without visible obstruction on physical examination. Blood pressure 140/90 mmHg, central venous pressure not increased. Arterial blood gas: pH 7.10, pCO2 77 mmHg, bicarbonate 24 mmol/L, and pO2 75 mmHg (on oxygen supplement). X-ray: massive pulmonary edema. Electrocardiogram: mild ST-elevation V2. Laboratory results: N-terminal pro b-type natriuretic peptide (NT-proBNP) 9.5pmol/L (normal <12pmol/L), high-sensitive troponin 440ng/L (ref.range 0-14ng/L). Furosemide was administered and Bilevel Positive Airway Pressure (BiPAP) was initiated but not tolerated. Echocardiography demonstrated basal hypercontractility and apical dyskinesia, suspicious for TTC.

Course: Due to progression to respiratory failure, mechanical ventilation was started. During intubation, a mobile cystic lesion was observed just above the vocal cords. Tracheal tube positioning was uneventful. Hemodynamic support was not required. After removing the cystic lesion, the patient was successfully extubated. Echocardiography showed normalisation of dyskinesia.

Pathology (cystic lesion): oncocytic cystadenoma.

Discussion: Management of massive pulmonary edema typically involves positive pressure ventilation. Nevertheless, our patient showed an adverse response to BiPAP. The underlying cause became evident during endotracheal intubation. The supposed mechanism is acute glottis closure by the mobile supraglottic structure during inspiration provoking NPPE and an additional adrenergic boost resulting in TTC, contributing to pulmonary edema. The therapeutic regimen is twofold: obstruction removal and cardiac support. NPPE is rare (incidence 0.05%-0.1%) and is post-anaesthesia related to laryngospasm. Combination of NPPE and TTC is even rarer and mostly seen in choking adults. Usually NT-proBNP is more elevated than troponin. Coronary obstruction must be excluded. Cardiac support is often needed with an expected full recovery.

Conclusion: NPPE caused by supraglottis obstruction provoking TTC with concomitant edema.

Take home message: Differentiate cardiac from non-cardiac pulmonary edema, but be aware of exceptional combinations needing a twofold therapeutic approach.

Literature:

1. Negative-Pressure Pulmonary Edema and Takotsubo Cardiomyopathy in the older Sakamoto T, et.al. Cureus 2022
2. Concurrent Negative-Pressure Pulmonary Edema (NPPE) and Takotsubo Syndrome (TTS) after Upper Airway Harmon E, et.al. Case Rep. Cardiology 2019

EP20

Veno-pulmonary ECMO as a bridge to recovery in refractory right ventricular failure after CABG

1. Teeuwen1, F.S. van den Brink1, J.A. Janson1, J.J. Maas1, J.E. Lopez Matta1, J.M. Montero Cabezas2, E.A.F. Mahtab3, C.V. Elzo Kraemer1

• Department of Intensive Care, Leiden University Medical Center, Leiden, the Netherlands
• Department of Interventional Cardiology, Leids Universitair Medisch Centrum, Leiden, the Netherlands
• Department of Thorax Surgery, Leiden University Medical Center, Leiden, the Netherlands

Teaser Case report

Een 58-jarige patiënte ontwikkelde ernstig rechter-ventrikelfalen na een coronair omleidingsoperatie, waarvoor behandeling met veno-arteriële extracorporele membraanoxygenatie (V-A ECMO) werd gestart. Herhaaldelijk weanen van V-A ECMO faalde, waarna werd besloten de configuratie te veranderen naar veno-pulmonaal. Onder geoptimaliseerde respiratoire en hemodynamische omstandigheden en na overweging van een bi-directionele cavo-pulmonaire anastomose als back-up strategie werd patiënte succesvol van ECMO geweaned en gedecanuleerd.

Case report

A 58-year-old female with a history of myocardial infarction and chronic kidney disease (CKD) underwent coronary artery bypass grafting (CABG) surgery in a referring hospital to treat anginous complaints. Postoperatively, acute right coronary artery occlusion caused cardiac arrest, and was treated by percutaneous coronary intervention. Trans-oesophageal ultrasound revealed severe right ventricular (RV) akinesia and impaired left ventricular function.

Haemodynamic deterioration prompted initiation of veno-arterial extracorporeal membrane oxygenation (V-A ECMO), with surgical cannulation of the subclavian artery and the right femoral vein. Continuous veno-venous hemofiltration was started to treat CKD progression. Repeated attempts to wean from V-A ECMO failed due to persistent RV dysfunction. After rejection for cardiac transplantation, the patient was transferred to our centre for advanced ECMO management.

On admission, the ECMO blood flow was 2.5 L/min. Norepinephrine was continued, and milrinone was stopped. A further weaning attempt failed two days later. After optimisation of respiratory and haemodynamic conditions, and surgical removal of small pericardial thrombi, the ECMO configuration was converted from V-A to veno-pulmonary (V-P) using a right internal jugular dual-lumen cannula (ProtekDuo™, LivaNova, USA). The ECMO blood flow was increased to 3.0 L/min to reduce RV load. Levosimendan (0.1 µg/kg/h in 24 hours) was administered to enhance cardiac contractility. Unfortunately, endotracheal extubation resulted in respiratory failure and re-intubation within 24 hours because of muscular weakness and sputum retention. Patient mobilisation was attempted, but was unsuccessful.

After ten days on V-P ECMO, a stepwise weaning strategy was started, reducing flow by 0.5 L/min per day to 1.5 L/min. Milrinone (0.25 µg/kg/h) was reintroduced 24 hours prior to another weaning trial. Bidirectional cavopulmonary anastomosis (Glenn Shunt) was considered as rescue strategy in case of failure (1). A weaning trial at 0.75 L/min was successful, despite minimal echocardiographic improvement of RV function. ECMO decannulation was completed uneventfully. Post-decannulation haemodynamics were favourable, with pulmonary vascular resistance of 1.8 Wood units, confirming low pulmonary vascular load, and potentiality for Glenn Shunt. The patient was weaned from mechanical ventilation after tracheostomy and transferred back to the referring hospital.

Concluding, this case demonstrates that conversion from V-A to V-P ECMO, combined with targeted inotropic optimisation, can facilitate successful weaning in patients with refractory RV failure. In selected cases, bidirectional cavopulmonary anastomosis may be considered as a rescue option when conventional weaning strategies fail.

References

M.C. den Haan, M. Palmen, A.D. Egorova, M.G. Hazekamp. (2024). Glenn shunt as a rescue strategy for acute right ventricular failure after right ventricular myocardial infarction. European Journal of Cardio-Thoracic Surgery, 65(5). https://doi.org/10.1093/ejcts/ezae157