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1.
BMJ Case Rep ; 17(7)2024 Jul 11.
Artículo en Inglés | MEDLINE | ID: mdl-38991576

RESUMEN

Systemic capillary leak syndrome (SCLS) is a rare and life-threatening disorder characterised by leaking of intravascular fluid to extravascular tissues. An association with immunotherapy and COVID-19 vaccination has been reported as potential triggers. A case of a patient in her 70s developing SCLS after the BNT162b2 (Pfizer-BioNTech) COVID-19 vaccination with a history of metastatic melanoma treated with nivolumab (PD-1 monoclonal antibody) and ipilimumab (anti-CTLA4 monoclonal antibody) is reported. The aetiology and management of SCLS are also reviewed in this case context.


Asunto(s)
COVID-19 , Síndrome de Fuga Capilar , Ipilimumab , Melanoma , Nivolumab , Humanos , Melanoma/tratamiento farmacológico , Síndrome de Fuga Capilar/inducido químicamente , Nivolumab/efectos adversos , Femenino , Ipilimumab/efectos adversos , Anciano , COVID-19/complicaciones , Vacunas contra la COVID-19/efectos adversos , Vacuna BNT162/efectos adversos , SARS-CoV-2 , Inmunoterapia/efectos adversos , Inmunoterapia/métodos , Neoplasias Cutáneas/tratamiento farmacológico , Antineoplásicos Inmunológicos/efectos adversos , Antineoplásicos Inmunológicos/uso terapéutico
3.
BMJ Case Rep ; 17(3)2024 Mar 18.
Artículo en Inglés | MEDLINE | ID: mdl-38499351

RESUMEN

Pythiosis is caused due to a filamentous eukaryotic micro-organism called Pythium insidiosum and the disease occurs commonly in horses and cattle. Subcutaneous pythiosis infection in humans is rare with no clear clinical guidelines for treatment. We present a case of a man in his 20s with non-resolving ulcers noted over lower extremity after exposure to swamp water draining animal remains. The patient received several courses of oral antibiotics with no improvement in symptoms before getting admitted to our institution. A diagnosis of subcutaneous pythiosis was made after deep wound culture following debridement detected P. insidiosum by use of PCR. Due to the rare incidence of such infection in humans and no clear guidelines available for treatment, the case was discussed with infectious disease specialists outside our institution and with veterinary physicians. An emergent approval for use of immunotherapy in conjunction with surgical debridement and antimicrobials was obtained from Food and Drug administration. The patient underwent successful treatment of infection and skin graft following treatment.


Asunto(s)
Antiinfecciosos , Pitiosis , Pythium , Masculino , Humanos , Animales , Caballos , Bovinos , Pitiosis/diagnóstico , Pitiosis/terapia , Desbridamiento , Inmunoterapia , Extremidad Inferior
4.
BMJ Case Rep ; 17(3)2024 Mar 29.
Artículo en Inglés | MEDLINE | ID: mdl-38553024

RESUMEN

A woman in her late 50s on mycophenolate for limited systemic sclerosis presented with abdominal pain. Vital signs and investigative evaluations were normal. Cross-sectional imaging identified gastric and small bowel wall thickening, free fluid, and pneumoperitoneum. In the operating room, a small bowel perforation was found and resected. Postoperatively, immunosuppression was held and she completed a course of amoxicillin/clavulanate. She discharged home and re-presented on postoperative day 8 with seizures and was found to have a frontal brain mass which was biopsied. Pathology from both the resected bowel and brain biopsy demonstrated Epstein-Barr virus-positive B-cell lymphoproliferative disorder with polymorphic B-cell features. The patient's immunosuppression was discontinued, and she was enrolled in a clinical trial for chemotherapy. Lymphoproliferative disorder can present years after immunosuppression initiation with either spontaneous perforation or solid tumour. Pathological assessment determines treatment options. Heightened concern for atypical clinical presentations in immunosuppressed patients is always warranted.


Asunto(s)
Infecciones por Virus de Epstein-Barr , Síndromes de Inmunodeficiencia , Perforación Intestinal , Trastornos Linfoproliferativos , Femenino , Humanos , Infecciones por Virus de Epstein-Barr/complicaciones , Infecciones por Virus de Epstein-Barr/diagnóstico , Infecciones por Virus de Epstein-Barr/patología , Herpesvirus Humano 4 , Enfermedad Iatrogénica , Síndromes de Inmunodeficiencia/complicaciones , Perforación Intestinal/complicaciones , Trastornos Linfoproliferativos/tratamiento farmacológico , Persona de Mediana Edad
5.
BMJ Case Rep ; 17(2)2024 Feb 17.
Artículo en Inglés | MEDLINE | ID: mdl-38367991

RESUMEN

Nivolumab is a programmed death-1 receptor blocker within the family of medications called immune checkpoint inhibitors (ICIs). Although generally well tolerated, cases of immune-related adverse events (irAEs) have been reported. We present a case of a man being treated with nivolumab for renal cell carcinoma who presented to the emergency department with problems of headache, fever and disorientation. After extensive evaluation, a diagnosis of immunotherapy-induced aseptic meningitis was considered more probable than infectious. Due to stable clinical status, no treatment was initiated, and the patient's condition improved spontaneously. The patient was discharged home. To date, only a handful of prior cases of nivolumab-induced meningitis have been reported. Our case demonstrates that irAEs can occur years after the initiation of ICIs. This was a milder presentation of a neurological irAE that resolved spontaneously with watchful waiting, showing that irAEs are likely an evolving spectrum of disease for which clinicians should be aware.


Asunto(s)
Antineoplásicos Inmunológicos , Neoplasias Renales , Meningitis Aséptica , Masculino , Humanos , Nivolumab/efectos adversos , Antineoplásicos Inmunológicos/uso terapéutico , Meningitis Aséptica/inducido químicamente , Meningitis Aséptica/tratamiento farmacológico , Fiebre/tratamiento farmacológico , Neoplasias Renales/tratamiento farmacológico , Estudios Retrospectivos
6.
BMJ Case Rep ; 17(1)2024 Jan 12.
Artículo en Inglés | MEDLINE | ID: mdl-38216167

RESUMEN

We report a case of vaccine-induced Sweet syndrome in a female patient in her 50s presenting with fevers and a scattered red patchy rash on the lower limbs. Seven days prior, she had received the first dose of AstraZeneca ChAdOx1-S vaccine. A skin biopsy confirmed Sweet syndrome. She did not respond to high doses of prednisolone and required methotrexate therapy to induce remission. This is one of the first reports of Sweet syndrome caused by the ChAdOx1-S vaccine and provides further evidence for vaccine-induced dermatosis. This case demonstrates that methotrexate can induce remission in cases of Sweet syndrome resistant to corticosteroids. This report also describes an approach to the differential diagnosis of patients presenting with a rash, fever and malaise.


Asunto(s)
Exantema , Síndrome de Sweet , Humanos , Femenino , Síndrome de Sweet/inducido químicamente , Síndrome de Sweet/diagnóstico , Síndrome de Sweet/tratamiento farmacológico , Metotrexato/uso terapéutico , Piel/patología , Fiebre/etiología , ChAdOx1 nCoV-19 , Exantema/patología
7.
BMJ Case Rep ; 16(12)2023 Dec 09.
Artículo en Inglés | MEDLINE | ID: mdl-38087481

RESUMEN

The evolution of SARS-CoV-2 within immunocompromised hosts who fail to clear the virus over many months has been proposed as a route to the development of Variants of Concern (VoCs). We present a case of an immunocompromised male patient with a prolonged SARS-CoV-2 infection. During hospitalisation, 7 weeks after first diagnosis, his condition worsened to require continuous ventilation support. Resolution of symptoms was observed after convalescent plasma therapy. Whole genome sequencing of the virus showed Pango lineage B.1.221. Between the first sample and the second from bronchoalveolar lavage fluid 7 weeks later, we identified eight mutations, including minor variants, which could be used to estimate the chronology of mutations. This suggests an elevated mutation rate, in-host accumulation of mutations and further evidence for sources of VoCs. Prolonged SARS-CoV-2 infections in immunocompromised hosts increase the likelihood of hospital stays and morbidity, and also pose an increased risk to global public health.


Asunto(s)
COVID-19 , SARS-CoV-2 , Humanos , Masculino , SARS-CoV-2/genética , COVID-19/terapia , Sueroterapia para COVID-19 , Inmunización Pasiva , Secuenciación Completa del Genoma , Huésped Inmunocomprometido , Mutación , Genoma Viral
8.
BMJ Case Rep ; 16(11)2023 Nov 17.
Artículo en Inglés | MEDLINE | ID: mdl-37977842

RESUMEN

Small cell carcinoma neuroendocrine type (SCCNET) is a rare tumour of the head and neck. Due to its infrequency, a paucity of data exists on optimal treatment, and the current paradigm for advanced SCCNET mirrors that of extensive small cell lung cancer. Increasingly, the treatment for extrapulmonary small cell carcinomas like SCCNET has incorporated immune checkpoint inhibitors (ICIs), although the utility of ICIs is not fully understood. We present a case of stage IVC sinonasal SCCNET in a woman in her 90s, who experienced eyelid swelling and unintentional weight loss. After diagnostic work-up, she was treated with etoposide, carboplatin and atezolizumab with a complete response to therapy. The patient had one episode of inflammatory polyarthropathy which resolved with steroids but otherwise tolerated treatment well and is now living with an overall survival of greater than 27 months. This case highlights the long-term efficacy of combination ICIs and chemotherapy in the treatment of SCCNET.


Asunto(s)
Carcinoma Neuroendocrino , Carcinoma de Pulmón de Células no Pequeñas , Carcinoma de Células Pequeñas , Neoplasias Pulmonares , Carcinoma Pulmonar de Células Pequeñas , Femenino , Humanos , Neoplasias Pulmonares/patología , Carcinoma de Células Pequeñas/tratamiento farmacológico , Carcinoma de Pulmón de Células no Pequeñas/tratamiento farmacológico , Carboplatino/uso terapéutico , Carcinoma Neuroendocrino/tratamiento farmacológico
9.
BMJ Case Rep ; 16(9)2023 Sep 15.
Artículo en Inglés | MEDLINE | ID: mdl-37714558

RESUMEN

A nulligravid woman in her mid-20s with colchicine-intolerant, steroid-dependent recurrent idiopathic pericarditis was in remission for 2 years on anakinra monotherapy when she had an unplanned pregnancy. Due to very limited and conflicting data, European Alliance of Associations for Rheumatology and American College of Rheumatology guidelines are equivocal on the use of anakinra in pregnancy, emphasising an individualised approach. Anakinra was discontinued but a month later, in the second month of gestation, the patient had an acute pericarditis flare. A multidisciplinary, patient-centred discussion about the competing risks of infection, obstetric complications and fetal malformations with anakinra versus suboptimally managed recurrent acute pericarditis guided the patient to choosing improved symptom control. Chest pain resolved and CRP normalised after daily anakinra injections were resumed. In the second trimester, the patient had mild COVID-19 infection and streptococcal pharyngitis on anakinra. At 34 weeks gestation, the patient went into preterm labour and had a spontaneous vaginal delivery of a healthy neonate.


Asunto(s)
COVID-19 , Pericarditis , Femenino , Recién Nacido , Embarazo , Humanos , Proteína Antagonista del Receptor de Interleucina 1/efectos adversos , Corticoesteroides , Enfermedad Crónica , Colchicina/efectos adversos , Pericarditis/tratamiento farmacológico , Recurrencia
10.
BMJ Case Rep ; 16(8)2023 Aug 30.
Artículo en Inglés | MEDLINE | ID: mdl-37648281

RESUMEN

Idiopathic hypereosinophilic syndrome is characterised by the overproduction of eosinophils with tissue infiltration, leading to multiorgan dysfunction. Its heterogenous presentation makes the diagnosis challenging and easy to miss. A woman in her 70s was admitted with chest pain and shortness of breath. Diagnostic testing showed elevated cardiac enzymes, an ejection fraction of 45% and pericardial effusion. Pericardiocentesis helped her symptoms significantly. Cardiac catheterisation revealed patent coronary arteries. She was diagnosed with myopericarditis and discharged on non-steroidal anti-inflammatory drugs. She returned the following week with worsening chest pain, dyspnoea and diarrhoea. Chest imaging showed bilateral infiltrates. Diagnostic testing showed eosinophilic predominance in peripheral blood (59%), pericardial fluid (37%) and bronchoalveolar lavage (31%). After a negative infectious workup, she was started on glucocorticoids and responded favourably. She was discharged on steroids. Mepolizumab was initiated outpatient, and steroids were discontinued. Mepolizumab was discontinued after 2 years while monitoring her symptoms and eosinophil counts.


Asunto(s)
Taponamiento Cardíaco , Derrame Pericárdico , Femenino , Humanos , Taponamiento Cardíaco/diagnóstico , Taponamiento Cardíaco/etiología , Derrame Pericárdico/etiología , Antiinflamatorios no Esteroideos , Lavado Broncoalveolar , Dolor en el Pecho , Disnea/etiología
11.
BMJ Case Rep ; 16(8)2023 Aug 16.
Artículo en Inglés | MEDLINE | ID: mdl-37586752

RESUMEN

A woman in her mid-60s presented to the hospital due to a history of nausea, vomiting, shortness of breath, dyspnoea on exertion and polyuria. She was receiving medical therapy for advanced non-small cell lung cancer and recently initiated immune checkpoint inhibitor (ICI) immunotherapy. Investigations revealed lab results consistent with diabetic ketoacidosis (DKA), elevated cardiac biomarkers, multiple cardiac arrhythmias and reduced ejection fraction on transthoracic echocardiogram. Cardiac catheterisation showed non-obstructive coronary arteries.The patient was diagnosed with an ICI-associated myocarditis and type I diabetes due to recent initiation of the ICI durvalumab. She was treated with the institutional DKA protocol and received corticosteroid therapy for drug toxicity according to guidelines. She was discharged with marked improvement in symptoms. The patient had good recovery after discharge with further investigations showing improvement in her cardiac ejection fraction on cardiac MRI. She remains on medical therapy with an insulin regimen for diabetes management.


Asunto(s)
Carcinoma de Pulmón de Células no Pequeñas , Diabetes Mellitus Tipo 1 , Cetoacidosis Diabética , Neoplasias Pulmonares , Miocarditis , Femenino , Humanos , Carcinoma de Pulmón de Células no Pequeñas/tratamiento farmacológico , Carcinoma de Pulmón de Células no Pequeñas/complicaciones , Inhibidores de Puntos de Control Inmunológico/uso terapéutico , Miocarditis/complicaciones , Neoplasias Pulmonares/tratamiento farmacológico , Neoplasias Pulmonares/complicaciones , Diabetes Mellitus Tipo 1/inducido químicamente , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Diabetes Mellitus Tipo 1/complicaciones , Cetoacidosis Diabética/complicaciones
13.
BMJ Case Rep ; 16(6)2023 Jun 20.
Artículo en Inglés | MEDLINE | ID: mdl-37339826

RESUMEN

Pembrolizumab is an immune checkpoint inhibitor used in many cancer types, including genitourinary cancers. Although immunotherapies have dramatically changed the landscape of cancer treatment by providing an alternative to traditional chemotherapy, they have been associated with significant immune-related adverse events (IRAEs) with wide-ranging clinical manifestations. We present the case of an elderly woman on pembrolizumab for metastatic bladder cancer who developed cutaneous IRAE with lichenoid eruptions that responded to high-dose intravenous glucocorticoids.


Asunto(s)
Erupciones Liquenoides , Neoplasias , Femenino , Humanos , Anciano , Inhibidores de Puntos de Control Inmunológico/uso terapéutico , Anticuerpos Monoclonales Humanizados/uso terapéutico , Erupciones Liquenoides/inducido químicamente , Erupciones Liquenoides/tratamiento farmacológico , Neoplasias/tratamiento farmacológico
14.
BMJ Case Rep ; 16(3)2023 Mar 07.
Artículo en Inglés | MEDLINE | ID: mdl-36882264

RESUMEN

This case reports a woman in her 40s with a history of allergic reaction to shellfish and iodine who presented with tongue angioedema, difficulty breathing and chest tightness after receiving the first dose of the Pfizer-BioNTech (BNT162b2) COVID-19 vaccine. Her angioedema remained for 10 days post-exposure to the vaccine, requiring 3 days of epinephrine infusion. She was discharged with advice to avoid further mRNA vaccines. This case highlights the increasing awareness needed of polyethylene glycol (PEG) allergy and the protracted nature of her reaction. A firm conclusion cannot be reached based on a single case report. More research is needed to understand whether there is a causal relationship between the BNT162b2 vaccine and PEG allergy. Awareness regarding PEG allergy and the complexities associated with it is important and needs to be raised due to its prevalent use in diverse industries.


Asunto(s)
Angioedema , COVID-19 , Femenino , Humanos , Vacuna BNT162 , Vacunas contra la COVID-19/efectos adversos , Angioedema/etiología , Vacunas de ARNm , Polietilenglicoles
15.
BMJ Case Rep ; 15(12)2022 Dec 27.
Artículo en Inglés | MEDLINE | ID: mdl-36574993

RESUMEN

Neuromyelitis optica is an autoimmune demyelinating astrocytopathy of the central nervous system that primarily affects the optic nerve and spinal cord. It is considered a multifactorial disease associated with antibodies against aquaporin 4, with complement cascade activation and lymphocytic infiltration leading to axonal loss and causing significant morbidity and disability. In addition, cases of inflammatory diseases of the central nervous system have been described after vaccination against SARS-CoV-2, mainly acute disseminated encephalomyelitis. Also, a few cases of neuromyelitis optica spectrum disorder, mostly aquaporin 4+, have been reported. We describe a patient who developed symptoms suggestive of acute disseminated encephalomyelitis the next day after vaccination against SARS-CoV-2. Three months later, a longitudinally extensive transverse myelitis compatible with aquaporin 4+ neuromyelitis optica was successfully treated with an interleukin 6 inhibitor. There is no proven association and research is needed to establish whether optic neuromyelitis is related to vaccination; this is a single case report from which no conclusion can be drawn.


Asunto(s)
COVID-19 , Encefalomielitis Aguda Diseminada , Neuromielitis Óptica , Humanos , Neuromielitis Óptica/etiología , Neuromielitis Óptica/complicaciones , Acuaporina 4 , SARS-CoV-2 , Encefalomielitis Aguda Diseminada/complicaciones , Autoanticuerpos , COVID-19/prevención & control , COVID-19/complicaciones , Vacunación/efectos adversos
16.
BMJ Case Rep ; 15(12)2022 Dec 30.
Artículo en Inglés | MEDLINE | ID: mdl-36585044

RESUMEN

A woman in her mid 40s presented for breast imaging after 1 week of painful and enlarged right axillary lymphadenopathy. She denied history of fever, weight loss, night sweats fatigue, cat scratch or other trauma. She received the second dose of Pfizer COVID-19 vaccine 3 months previously on the contralateral arm. A mammogram demonstrated a single, asymmetric, large and dense right axillary lymph node. Ultrasound confirmed a 2.5 cm lymph node with cortical thickening of 0.6 cm. Ultrasound-guided core biopsy showed necrotising lymphadenitis with associated aggregates of histiocytes and plasmacytoid dendritic cells. Potential causes of necrotising adenitis including Bartonella, tuberculosis, Epstein-Barr Virus, herpes simplex virus, systemic lupus erythematosus and lymphoma were excluded. In the absence of any identifiable infectious or autoimmune causes, and given the temporal relatedness with vaccine administration, it was determined that the Kikuchi-Fujimoto-like necrotising lymphadenitis was likely secondary to the COVID-19 vaccine. To date, there has been no casual association made between the COVID-19 vaccine and KFD necrotising lymphadenitis.


Asunto(s)
Vacunas contra la COVID-19 , COVID-19 , Infecciones por Virus de Epstein-Barr , Linfadenitis Necrotizante Histiocítica , Linfadenitis , Linfadenopatía , Femenino , Humanos , COVID-19/complicaciones , Vacunas contra la COVID-19/efectos adversos , Infecciones por Virus de Epstein-Barr/complicaciones , Herpesvirus Humano 4 , Linfadenitis Necrotizante Histiocítica/etiología , Linfadenitis Necrotizante Histiocítica/complicaciones , Biopsia Guiada por Imagen/efectos adversos , Linfadenopatía/etiología
17.
BMJ Case Rep ; 15(12)2022 Dec 13.
Artículo en Inglés | MEDLINE | ID: mdl-36524267

RESUMEN

We present a female kidney transplant patient under conventional immunosuppression therapy. Her humoral immunity study (anti-spike-specific antibodies) was negative after the initial regimen and the third dose of vaccination against COVID-19. The specific ex vivo cellular immune study against spike of SARS-CoV-2 by interferon gamma release assay (IGRA) also remained at non-response levels at different time points despite an optimal non-specific cell immune response assessment. However, the cellular immunity test by delayed-type hypersensitivity (DTH) with spike of SARS-CoV-2 was always positive since the vaccination scheme began. Only after COVID-19 infection has there been a seroconversion of the patient's antibody tests along with IGRA positivity. The use of DTH test to measure the immune response could be a better and earlier parameter of the actual immune status that helps us to predict the immune response in real life. Hybrid immunity combining vaccine and natural infection could be a stronger stimulator of the specific global immune response.


Asunto(s)
COVID-19 , Trasplante de Riñón , Femenino , Humanos , SARS-CoV-2 , Pacientes , Vacunación , Inmunidad Humoral , Anticuerpos Antivirales , Inmunidad Celular
18.
Artículo en Inglés | MEDLINE | ID: mdl-36262897

RESUMEN

Multisystem inflammatory syndrome is a life-threatening condition associated with elevated inflammatory markers and multiple organ injury. A diagnosis of exclusion, it has been reported after severe acute respiratory syndrome coronavirus 2 infection (SARS-CoV-2) in children and adults; recently it has been described in some post-COVID-19 vaccinated individuals. The prognosis with supportive care and immunomodulatory therapy is good, although some individuals may require treatment in the intensive care unit (ICU). Here we report a case of a 58-year-old man who developed multi-organ failure after receiving the second dose of the Moderna mRNA-1273 COVID-19 vaccine. He required critical organ support in the ICU. An extensive workup was done to rule out alternative infectious and inflammatory processes. Following a period of gradual in-hospital convalescence, our patient made a full recovery. To our knowledge, this is the first comprehensively described case of multisystem inflammatory syndrome associated with Moderna mRNA-1273 COVID-19 vaccine in an adult over 50 years of age.

19.
BMJ Case Rep ; 15(8)2022 Aug 23.
Artículo en Inglés | MEDLINE | ID: mdl-35999022

RESUMEN

Tuberculosis (TB) remains a significant cause of morbidity and mortality globally. The disseminated form of the disease has a worse prognosis and is commonly associated with primary and acquired immunodeficiency states such as HIV/AIDS, post-organ transplant and malnutrition. However, disseminated TB in the context of isolated impaired cellular responses to interleukin (IL)-23 due to tyrosine kinase 2 (TYK2) deficiency has been rarely reported. We highlight the case of a young woman with pulmonary and central nervous system TB associated with previously undiagnosed IL-23/TYK2 signalling defects causing impaired response to IL-23. A significant clinical improvement was observed after introduction of adjunctive interferon-gamma therapy to her anti-tuberculous medications. This case emphasises the need to broadly evaluate for potential immune deficiencies in poorly responding patients with fully sensitive TB as well as the potential benefits of interferon-gamma therapy in patients with certain immune defects.


Asunto(s)
Mycobacterium tuberculosis , Tuberculosis del Sistema Nervioso Central , Tuberculosis Miliar , Femenino , Humanos , Interferón gamma/uso terapéutico , Interleucina-23 , TYK2 Quinasa
20.
BMJ Case Rep ; 15(8)2022 Aug 01.
Artículo en Inglés | MEDLINE | ID: mdl-35914801

RESUMEN

Daratumumab-containing regimens are an effective treatment for advanced cases of multiple myeloma. Overall, daratumumab has a favourable safety profile, although rare side effects can occur. Rare side effects of daratumumab include choroidal effusion. Patients who begin to experience symptoms such as eye swelling, vision changes, eye discharge and blurry vision should undergo urgent ophthalmological evaluation and their daratumumab infusions held.


Asunto(s)
Efusiones Coroideas , Mieloma Múltiple , Anticuerpos Monoclonales/uso terapéutico , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Humanos , Mieloma Múltiple/terapia , Resultado del Tratamiento
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