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BACKGROUND Catastrophic antiphospholipid syndrome (CAPS) is a rare, life-threatening form of antiphospholipid syndrome characterized by widespread thrombotic complications leading to multiorgan ischemia and failure. Although there are no standard treatment guidelines for CAPS, it often involves triple therapy with anticoagulation, corticosteroids, and plasma exchange. Recently, biologics such as rituximab and eculizumab have also shown promise as potential new therapies for CAPS, as observed in our case. CASE REPORT We describe a 59-year-old female patient who presented with altered mental status and diffuse weakness. Imaging studies revealed multiorgan thrombosis along with thrombocytopenia that markedly improved with plasma exchange therapy, steroids, and a heparin drip. While the exact etiology of CAPS remained unknown, it was likely precipitated by her warfarin discontinuation and confirmed Haemophilus influenzae infection. The patient's hospital course was complicated by hemorrhagic shock after a renal biopsy, followed by an acute drop in thrombocytopenia and new embolic infarcts in the brain that raised concern for CAPS re-emergence. To address the refractory nature of her condition, the patient underwent a trial of rituximab, which remarkably improved her clinical picture and platelet count by an 8-fold increase within 1 week. CONCLUSIONS This case highlights the importance of early recognition and diagnosis of catastrophic antiphospholipid syndrome, a true rheumatological emergency that requires aggressive treatment to prevent irreversible complications. Our patient's presentation and response to treatment also underscores the complexity of managing CAPS and the use of newer biological therapies in refractory cases.
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Síndrome Antifosfolípido , Trombocitopenia , Trombosis , Femenino , Humanos , Persona de Mediana Edad , Síndrome Antifosfolípido/complicaciones , Síndrome Antifosfolípido/diagnóstico , Rituximab/uso terapéutico , Trombosis/etiología , Heparina/uso terapéutico , Trombocitopenia/complicacionesRESUMEN
We present a novel case of severe hyperosmolar hyperglycemic derangement in an elderly patient - without a known history of diabetes mellitus - after the first injection of leuprolide for the treatment of metastatic prostate adenocarcinoma. Whilst the available literature provided accumulative evidence of an association between insulin resistance and the use of gonadotropin-releasing hormone (GnRH) agonists, the initial presentation of leuprolide-induced impaired glycemic tolerance with a hyperosmolar hyperglycemic state (HHS) represents a clinical rarity that was seldom reported. A literature review was conducted to explore the underlying mechanisms of leuprolide-associated glucose intolerance. Screening for diabetes is recommended for patients receiving leuprolide therapy to identify at-risk patients and close glycemic monitoring is warranted in diabetic patients to minimize serious complications from poor glycemic control induced by leuprolide.
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Kaposi sarcoma (KS) is caused by human herpesvirus 8 (HHV-8). Epidemic KS is described in the human immunodeficiency virus (HIV) population with acquired immune deficiency syndrome (AIDS). It primarily affects the skin, but it may uncommonly disseminate to involve extracutaneous sites such as the gastrointestinal (GI) tract, liver, and lungs. In this case report, the authors report a 26-year-old homosexual male who was admitted with acute hypoxemic respiratory failure. He was diagnosed with an HIV infection about five months before index presentation, and he was commenced on highly active antiretroviral therapy (HAART). Physical examination was remarkable for diffuse cutaneous nodules over the lower extremities, back, and oropharynx. Chest imaging revealed diffuse bilateral infiltrates, mediastinal adenopathy, and a persistent bilateral pleural effusion. Extensive diagnostic workup was negative for underlying infectious etiology. Transbronchial biopsy demonstrated proliferated spindle cells that stained positive for HHV-8 in keeping with pulmonary KS. Skin biopsies also concurred with the diagnosis of cutaneous KS. Interestingly, the cluster of differentiation 4 (CD4) count was 647 cells/mm3, and HIV viral load (VL) was 500 copies/ml. This case demonstrated an atypical natural history of pulmonary KS in an HIV patient as pulmonary and disseminated mucocutaneous KS occurred with a relatively higher CD4 count (≥500 cells/mm3). It also reminds pulmonologists and infectious disease specialists to consider pulmonary KS as a differential diagnosis of acute hypoxemic respiratory failure in HIV patients, even in the absence of other clinical and laboratory criteria that define the AIDS stage.
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Actinomycosis is a chronic inflammatory infectious disease that can affect various organ systems. Pulmonary actinomycosis is an exceptionally uncommon clinical occurrence that yet deserves special attention, as it closely mimics a broad spectrum of infectious and neoplastic lung pathologies. The non-specific nature of its clinical features and radiological appearances makes early diagnosis quite challenging. The authors reported a 25-year-female with poorly controlled diabetes mellitus and morbid obesity who presented with a one-week history of unilateral, right-sided, pleuritic chest pain and shortness of breath. Chest imaging revealed a suspicious right hilar soft tissue mass encasing the right upper lobe bronchus with post-obstructive atelectasis. Transbronchial biopsy revealed suppurative granulomatous inflammation, and anaerobic cultures from the bronchial tissues grew Actinomyces species that were identified using the matrix-assisted laser desorption/ionization-time of flight (MALDI-TOF) technique. A long course of penicillin-based antibiotics was employed, and follow-up imaging revealed a satisfactory response to the antimicrobial therapy. This case demonstrates that microbiological examination is imperative to accurately diagnose the etiology of suspicious lung masses in young immunocompromised hosts. It also proves the diagnostic value of the MALDI-TOF technique in the early identification of Actinomyces species.
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Venous thromboembolism (VTE) is a recognized extraintestinal manifestation of inflammatory bowel disease (IBD), with deep venous thrombosis (DVT) and pulmonary embolism being reported as the most frequent vascular complications in IBD patients. Much less frequently, arterial thromboembolic events may also be associated with greater morbidity and mortality. Aortic mural thrombosis is a rare phenomenon described in patients with IBD that often results in serious consequences such as visceral infarction and acute ischemia of the lower extremities. We described an unusual case of a female patient with Crohn's disease (CD) who presented with generalized abdominal pain and vomiting. Imaging showed an active flare-up of intestinal CD as well as two mural thrombi in the distal descending thoracic aorta and the abdominal aorta at the level of the left renal artery, respectively, with a left renal infarction. The mesenteric angiogram revealed a patent celiac axis and mesenteric arteries. The patient was therapeutically anticoagulated, and she underwent a right hemicolectomy for the perforated ileal disease. A comprehensive diagnostic workup for hypercoagulability and thrombophilia was negative for an underlying etiology, and the active CD flare-up was considered the main culprit triggering the aortic thrombosis in this reported patient. Our case highlighted the occurrence of aortic thrombosis in a patient with IBD and that entails careful attention. Early recognition and timely management with a multidisciplinary team is the key to improving the outcome of aortic events that coincide with the active flare-up of IBD.
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Hematogenous spread is fairly an unusual feature for papillary thyroid carcinoma (PTC) in comparison to follicular thyroid carcinoma (FTC). Thoracic spinal metastasis with complicating cord compression is an even rarer manifestation of PTC that was reported in a limited number of cases in the literature. Herein we present a 65-year-old female with a history of PTC on current radiotherapy, status post attempted surgery due to significant tumor burden and intraoperative bleeding, presented with a one-week history of rapidly progressive bilateral lower extremities weakness. Physical examination revealed paraplegia of both lower extremities with areflexia and a sensory level equivalent to the upper thoracic vertebrae. Urgent imaging depicted destructive epidural lesions at T1-T3 vertebrae with thoracic cord compression. Emergent laminectomy and debulking of these lesions were undertaken. Histopathological examination confirmed metastatic PTC. The patient proceeded to further treatment with radiotherapy following her successful neurological recovery. Thoracic vertebral metastasis is an unusual oncological phenomenon of PTC. Metastatic PTC should be considered in patients with a current or remote history of PTC who present with thoracic cord compression. Our case demonstrates that multidisciplinary management is the key to achieving a better outcome for metastatic PTC with thoracic cord compression.
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Tocilizumab is a recombinant humanized monoclonal antibody directed against the interleukin-6 (IL-6) receptor, which has been used for the treatment of rheumatoid arthritis (RA). A range of side effects have been associated with tocilizumab, with gastrointestinal perforation (GIP) being described as a rare but potentially life-threatening complication that deserves considerable attention. The authors report a case of a young male patient with a history of challenging RA who encountered a lower GIP that was associated with tocilizumab therapy. The occurrence of tocilizumab-induced GIP in this reported patient had initially posed a diagnostic dilemma, as its clinical presentation mimicked other autoimmune inflammatory and infectious diseases that are commonly associated with RA. Physicians should be aware of GIPs as a serious adverse event of tocilizumab use despite being a rare phenomenon, particularly in the era of the global pandemic of coronavirus disease 2019 (COVID-19), when this novel drug has been authorized for the management of selected patients with severe COVID-19 infection. Therefore, early recognition and timely management of GIPs would minimize potential morbidities associated with critically ill COVID-19 patients.
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Acute abdomen is a common emergency condition affecting young adults, and the first consideration is usually aimed to rule out acute appendicitis in this age group. Omental fat torsion has emerged as one of the rare etiologies of acute abdomen in the younger population. It warrants serious consideration as it closely mimics acute appendicitis in its clinical presentation. Herein we report a case of omental fat torsion in a 22-year-old male patient who presented with an acute right-sided lower abdominal pain which was highly suggestive of acute appendicitis. However, the diagnostic laparoscopy revealed a normally looking appendix and terminal ileum with an infarcted omental segment on the right side of the greater omentum. A laparoscopic omentectomy and an appendectomy were performed with an uneventful postoperative recovery. The pathology report confirmed omental fat infarction and a normal appendix. This case highlights omental fat infarction as a rare etiology of acute abdomen in a young male patient.
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Abdomen Agudo , Traumatismos Abdominales , Apendicitis , Enfermedades Peritoneales , Abdomen Agudo/etiología , Traumatismos Abdominales/complicaciones , Enfermedad Aguda , Adulto , Apendicitis/complicaciones , Apendicitis/diagnóstico , Apendicitis/cirugía , Humanos , Infarto/complicaciones , Infarto/patología , Infarto/cirugía , Masculino , Epiplón/patología , Epiplón/cirugía , Enfermedades Peritoneales/diagnóstico , Enfermedades Peritoneales/etiología , Enfermedades Peritoneales/cirugía , Anomalía Torsional/complicaciones , Anomalía Torsional/diagnóstico , Anomalía Torsional/cirugía , Adulto JovenRESUMEN
BACKGROUND Sarcoidosis is a systemic granulomatous disease which predominantly affects the lungs, skin, and lymph nodes. Vertebral sarcoidosis is a rare entity. The clinical presentation of sarcoidosis with diffuse vertebral osseous and visceral lesions, simulating a disseminated metastatic cancer, is extremely unusual and has been reported only in a handful of cases in the current literature. CASE REPORT A 78-year-old White female patient with a remote history of asymptomatic pulmonary sarcoidosis presented with a 1-month history of generalized weakness. Physical examination was positive for upper and lower midline spinal tenderness. Laboratory findings showed anemia, hypercalcemia, and deranged liver functions. Abdominal imaging revealed an enlarged and nodular liver, ascites, splenomegaly, and enlarged retroperitoneal lymph nodes. Spinal imaging demonstrated several multi-level vertebral osseous lesions suspicious for metastatic bone cancer. Following extensive diagnostic work-up to rule out underlying metastatic cancers, a bone biopsy from an iliac lesion demonstrated active non-caseating granulomas, and a diagnosis of multisystemic sarcoidosis was made. The patient was started on systemic corticosteroids and demonstrated a gradual symptomatic improvement. Follow-up imaging revealed interval resolution of vertebral lesions. CONCLUSIONS The clinical and radiological features of vertebral sarcoidosis can be indistinguishable from metastatic bone cancers. The possibility of widespread extrapulmonary sarcoidosis should be considered in any patients with a remote history of pulmonary sarcoidosis who experience simultaneous onset of unexplained multisystemic symptoms.