2023: Vol 10 No 6

2023: Vol 10 No 6
  • Syed Aun Muhammad, Fiona Coath, James Jegard
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    Introduction: We describe a case of remitting seronegative symmetrical synovitis with pitting oedema (RS3PE) syndrome following administration of the ChAdOx1-S/nCoV-19 [recombinant] vaccine, suggesting a possible causal link.
    Case Description: A 72-year-old man presented to his general practitioner with swollen, oedematous hands and legs 2 weeks after receiving a coronavirus vaccine. He had raised inflammatory markers but remained systemically well. He was initially presumed to have cellulitis, but his symptoms persisted despite several courses of antibiotics. Deep vein thromboses, cardiac failure, renal failure and hypoalbuminaemia were ruled out. Upon Rheumatology review, he was diagnosed as having RS3PE syndrome with the Covid vaccine suspected of being an immunogenic trigger. Following initiation of steroid therapy, his symptoms improved dramatically, as is characteristic of RS3PE syndrome.
    Discussion: The pathophysiology of RS3PE is unclear. It is known to have various triggers and associations including infections, certain vaccines and malignancy. This case highlights that a coronavirus vaccine (ChAdOx1-S/nCoV-19 [recombinant] vaccine) is also a possible trigger. Factors that make the diagnosis likely include an acute onset of symptoms including pitting oedema in a typical distribution, age above 50, and unremarkable autoimmune serology. Other learning points from this case include the importance of antibiotic stewardship and the need to explore non-infectious causes of illness when antibiotics do not improve symptoms.
    Conclusion: The ChAdOx1-S/nCoV-19 [recombinant] vaccine is a possible trigger of RS3PE. However, the benefits of vaccines against coronavirus outweigh the risks in the majority of patients.

  • Joana Bernardino Cardoso, Ana Silveira, Sara Duarte, Ruth Fortes, Ahmed Botelho, Ana Simas, Rui Suzano
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    Pseudopheochromocytoma is a pathological condition presenting with paroxysmal hypertension with normal or moderate elevation in catecholamines and metanephrine levels, but no evidence of a tumoural cause. Imaging studies and I-123 metaiodobenzylguanidine scintigraphy are essential for exclusion of pheocromocytoma.
    We describe a case of pseudopheochromocytoma related to levodopa in a patient with paroxysmal hypertension, headache, sweating, palpitations and increased plasmatic and urinary metanephrine levels, without adrenal or extra-adrenal tumour. The beginning of the patient´s clinical symptoms coincided with the initiation of the levodopa treatment and the complete resolution of the symptoms occurred after the discontinuation of levodopa.

  • Farah Heis, Mohamed Heis, Mahrukh Khan, Manjula Ashok
    Views: 393 HTML: 56 PDF: 361

    There has been a recent outbreak of monkeypox in the United States, mostly among men who have sex with men (MSM). It seems to be a self-limiting disease but can be potentially serious in immunocompromised patients. Monkeypox infection is transmitted mainly through skin-to-skin contact and possibly seminal and vaginal fluids. Very few cases of monkeypox infection in immunocompromised patients have been published in the literature. We report a case of infection in a renal transplant recipient, and describe the clinical course and outcome.

  • Mårten Söderberg, Erika Smedberg, Pelle G. Lindqvist
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    Introduction: Pulmonary embolism (PE) is a rare, severe complication in pregnancy, in which case thrombolysis can be lifesaving but has risks. We aim to highlight actions specific to pregnant women.
    Case Description: A 24-week pregnant woman developed shortness of breath and experienced sudden cardiac arrest. Cardiopulmonary resuscitation (CPR) was begun immediately in the ambulance and a perimortem caesarean section was performed upon arrival at hospital, but the new-born died. After 55 minutes of CPR, bedside echocardiography revealed right ventricular strain and thrombolysis was given. The uterus was bandaged to minimize blood loss. After massive transfusions and correction of haemostasis, a hysterectomy was performed due to inability of the uterus to contract. After 3 weeks, the patient was discharged in good health and placed on continuous anticoagulant treatment with warfarin.
    Discussion: Approximately 3% of all out-of-hospital cardiac arrest cases are due to PE. Among the few patients who survive at the scene, thrombolysis can be lifesaving and should be considered in pregnant women with unstable PE. Prompt collaborative diagnostic work-up in the emergency room is necessary. In a pregnant woman with cardiac arrest, a perimortem caesarean section improves the chances of both maternal and fetal survival.
    Conclusion: Thrombolysis should be considered for patients with PE in pregnancy with the same indications as in a non-pregnant woman. In case of survival, there is profuse bleeding with need for massive transfusions and haemostasis correction. Despite being in very poor condition, the above patient survived and was fully restored to health.

  • Álex García Tellado, Carmen Lasa Teja, María Rodríguez Vidriales, Mercedes De la Fuente Vázquez, Patricia Marín Oliván, Francisco Arnaiz Las Revillas Almajano
    Views: 344 HTML: 61 PDF: 325

    Introduction/Aims/Background: Pyoderma gangrenosum is an immune-mediated illness that can be caused by several affections, such as inflammatory bowel disease, rheumatoid arthritis, and drug use. We present a rare case of pyoderma gangrenosum induced by levamisole-adulterated cocaine. There have been few cases of this disease reported in the world. Levamisole is an anthelmintic drug used to adulterate cocaine to boost its effect. It also has immune-modulating effects causing, among others, vasculitis and dermatological problems.
    Materials and Methods: Clinical case of a 46-year-old man admitted to the hospital University Marqués de Valdecilla in Santander, Spain, in August 2022. We diagnosed pyoderma gangrenosum based on clinical, analytical, and histological parameters.
    Results: We report a case of pyoderma gangrenosum induced by consumption of levamisole-adulterated cocaine.
    Discussion: This patient suffered from a rare and extensive immune-mediated affection with characteristic primary lesions in the form of suppurative ulcers that responded to immunosuppressive treatment. Behind pyoderma gangrenosum there may be underlying conditions such as inflammatory bowel disease, or pyoderma gangrenosum may be secondary to identifiable causes such as cocaine use as in this patient.

  • Paul Silaghi, Daniel T. Eitzman
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    Sickle cell disease is a prevalent hematologic condition, but some of the factors that lead to erythrocyte sickling are not fully known. A 58-year-old male patient with a history of sickle cell disease (SCD) and paroxysmal atrial fibrillation was transferred from an outside hospital for further management of refractory sickle cell crisis with acute chest syndrome. Before transfer, the patient received antibiotics and multiple packed red blood cell (pRBC) transfusions, with minimal effect on symptoms or anemia. After transfer, the patient developed rapid supraventricular tachycardia and atrial fibrillation (rates >160) with a drop in blood pressure. He was started on IV amiodarone. His heart rate was subsequently better controlled and converted to sinus rhythm the following day. Three days following initiation of amiodarone, the patient, with a hemoglobin count of 6.4 g/dl, required one additional unit of pRBC. On the fourth day, the patient’s hemoglobin count rose to 9.4 g/dl, and he reported a marked improvement in symptoms. The improvements in symptoms and hemoglobin count were sustained, and the patient was discharged two days later. This remarkable improvement in anemia and symptoms triggered a search for potential causes. Amiodarone is a complex drug shown to have effects on multiple cell types, including erythrocytes. A recent preclinical study demonstrated reduced sickling and improved anemia in a murine model of SCD. This case report raises the possibility that amiodarone may have contributed to the rapid improvement in anemia and should be further explored in clinical trials.

  • Ilir Tolaj, Visar Gashi, Gramoz Bunjaku, Murat Mehmeti, Hajrullah Fejza
    Views: 461 PDF: 359 HTML: 379

    Background: Candida cellulitis is a rare disease, primarily reported in immunocompromised patients. Atypical Candida spp. infections are increasing, largely due to the growing number of immunocompromised patients. This case report describes a 52-year-old immunocompetent patient with facial cellulitis caused by Candida guilliermondiCandida guilliermondi has not previously been reported as a cause of facial cellulitis in either immunocompromised or immunocompetent patients.
    Case presentation: A 52-year-old, otherwise healthy, male patient presented with facial cellulitis that did not respond to intravenous antibiotics. Culture of the drained pus revealed Candida guilliermondi. The patient was successfully treated with intravenous fluconazole.
    Conclusion: This case highlights the possibility of atypical Candida spp. causing deep facial infections in immunocompetent patients. Candida guilliermondi has not been previously reported as a cause of facial cellulitis in either immunocompromised or immunocompetent patients. Healthcare providers should consider atypical Candida spp. infections in the differential diagnosis of deep facial infections in both immunocompromised and immunocompetent patients.

  • Pierre Rossignon, Alain Soupart
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    Adult-onset Still’s disease (AOSD) is a rare autoinflammatory disorder that can lead to a cytokine storm, causing a range of symptoms. Acute intestinal pseudo-obstruction is another rare condition that results in intestinal obstruction without anatomical cause. Although the two conditions are rarely reported together, we present the case of a 62-year-old male who developed acute intestinal pseudo-obstruction in the context of an AOSD flare. This led to severe hypokalaemia and a critical condition. Other symptoms included a high-spiking fever lasting for weeks, polyarthralgias and a typical salmon-coloured rash. After ruling out other potential causes, the patient was diagnosed with AOSD. Our findings suggest that the cytokine storm associated with this disease triggered the acute intestinal pseudo-obstruction and life-threatening hypokalaemia, establishing a causal relationship.
    Only four other cases of AOSD complicated by intestinal pseudo-obstruction have been reported, and this is the first to present with life-threatening hypokalaemia. This case serves as a crucial reminder that, despite being a diagnosis of exclusion, Still’s disease should be considered as a potential cause of intestinal pseudo-obstruction, as prompt recognition and treatment of the underlying cause is crucial in managing this potentially life-threatening condition.

  • Gaurav Mohan, Ashika Ajitkumar, Sivanaga Yarrarapu, Karthik Ashok, Doantrang Du, Douglas Livornese
    Views: 385 HTML: 64 PDF: 328

    Tracheoesophageal prosthesis (TEP) is an artificial connection between the trachea and esophagus allowing air into the upper esophagus from the trachea thereby vibrating it. TEPs give patients who lose their vocal cords to laryngectomies a tracheoesophageal voice. A potential complication of this is silent aspiration of gastric content. We present a case of a 69-year-old female with a TEP placed after a laryngectomy for laryngeal cancer who presented to the hospital with shortness of breath and hypoxia. She was initially treated for a presumed diagnosis of chronic obstructive pulmonary disease (COPD) and congestive heart failure (CHF) exacerbations but continued to be hypoxic despite aggressive medical management. Further evaluation revealed silent aspirations as a consequence of TEP malfunction. Through our case report we urge clinicians to consider this differential diagnosis, as the clinical presentation of silent aspiration among patients with a TEP can be easily mistaken for a COPD exacerbation. A large number of patients with TEPs are smokers with underlying COPD.

  • Ariana Maia, Catarina Cidade Rodrigues, Isabel Ribeiro, Cláudia Amaral
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    We report a case of a 19-year-old young male presenting with thyrotoxicosis with inappropriately elevated TSH. Magnetic resonance imaging revealed a pituitary adenoma (8.2 x 9.7 mm), TRH stimulation test showed abnormal blunted TSH response, and serum glycoprotein hormone alpha-sub-unit was elevated. He had no family history of thyroid disease and TR-beta genetic testing excluded resistance to thyroid hormone action. The diagnosis of thyrotropin-secreting pituitary adenoma (TSHoma) was presumed and long-acting somatostatin analogue was promptly initiated. After two months of octreotide treatment, serum TSH and FT3 returned to within normal ranges. Tumour resection by transsphenoidal surgery was performed and, ten days after surgery, clinical hypothyroidism was achieved, despite detectable TSH levels (TSH 1.02 µU/ml[RR 0.27-4.2]). Although the patient remained euthyroid for the following three years, there was a gradual biochemical elevation in the levels of TSH, FT4, and FT3 over time, reaching serum values above the normal limit in the third year after surgery. Imaging did not show neoplasm recurrence at this point. After two years, the patient presented with clinical manifestations of re-onset thyrotoxicosis, with MRI revealing a T2 hypersignal oval area compatible with a pituitary adenoma. Adenectomy was performed. Histopathological and immunohistochemical analyses revealed a pituitary adenoma with transcription factor PIT1 expression and positivity for TSH and PRL. TSHoma treatment may not be always effective in the first therapeutic approach and recurrences are a possibility, making follow-up essential. The present case highlights the heterogeneity of post-treatment cure criteria and their limitations.

  • Léa Docquier, Xavier Jannot, Zaida Córdoba-Sosa, Léa Pierre, Pierre Alexis Austrusseau, Emmanuel Andres, Noel Lorenzo Villalba
    Views: 598 HTML: 53 PDF: 473

    We report on a 70-year-old male patient admitted to the internal medicine department for evaluation of a right cervical mass. He had been treated with antibiotics as an outpatient by his primary care doctor. Upon admission the patient was asymptomatic, but within a few hours his cervical mass enlarged; this enlargement was confined to the right sternocleidomastoid muscle. Complete blood investigations including serology and autoimmunity were negative. The neck scan and MRI were in favour of myositis. No other lesions were found either in the nasal fibre-optic exam or in the thoracic-abdominal-pelvic scan. The biopsy of the muscle showed a lymphoplasmacytic inflammatory infiltrate of the perimysium. The diagnosis of focal myositis was made. The patient clinically improved during hospitalisation with complete resolution of symptoms without any specific intervention.