Hiccups are common; however, hiccups caused by sarcoidosis have rarely been reported. An unusual case involving a patient with persistent hiccups possibly caused by hilar/mediastinal lymph node enlargement due to sarcoidosis prompted us to perform a literature search. Eight case reports relating hiccups to sarcoidosis were found and in only one case were the hiccups thought to be due to thoracic lymphadenopathy (LAD). Most cases were attributed to involvement of the central nervous system (CNS) with sarcoidosis. Management of hiccups in general is unclear and only chlorpromazine is approved by the Food and Drug Administration (FDA) for treatment; multiple other pharmacological agents have been advocated mostly being ineffective. This case report describes a patient whose hiccups were likely caused by thoracic sarcoidosis. It reviews the mechanisms of hiccups, explores co-morbid conditions associated with hiccups (including sarcoidosis), and provides some recommended treatments.
Respiratory complaints are common symptoms in pregnancy, and it is important to delineate whether the causative factor is cardiac, pulmonary, or physiologic in origin. The pathophysiology of Sickle Cell Disease (SCD) can further complicate the differential diagnosis. To our knowledge, there is little to no literature devoted to the management of pulmonary complications in the pregnant sickle cell patient. Therefore we present a review of the current literature and feature reports on the incidence and prevalence of the pulmonary complications in a sickle cell pregnancy. These include Acute Chest Syndrome (ACS), Pulmonary Hypertension (PH), Pulmonary Embolism (PE), and sickle cell chronic lung disease. We also aim to recommend management strategies that address the SCD-related pulmonary complications in the pregnant state.
Hemophagocytic lymphohistiocytosis (HLH) is a rare but life-threatening disease. It is characterized by abnormalities in immunoregulation due to a non-malignant proliferation of lymphocytes and secretion of inflammatory cytokines. HLH is typically associated with acquired immunodeficiencies such as in hematopoietic stem cell transplantation. We present a case of a young woman who was treated in our ICU for confirmed HLH which was attributed to an Epstein-Barr virus (EBV) infection that reactivated during her post-transplant immunosuppression.CASE PRESENTATION: A 46-year-old female presented to her primary care physician with gradual dyspnea, spontaneous gum bleeding and lower extremity bruising. She was diagnosed with anemia during her pregnancy. Eventually developed leukopenia and pancytopenia. She was later diagnosed with aplastic anemia and underwent a conditioning regimen prior to peripheral blood stem cell transplantation (PBSCT) with cyclophosphamide, fludarabine, total body irradiation, and rabbit anti-thymocyte globulin (ATG). She had an allogeneic PBSCT from a 10/10 HLA-matched unrelated donor (MUD). Her pre-transplant workup was positive EBV IgG. Donor pre-transplant workup was negative for EBV. Post-transplant immunosuppression consisted of rituximab and tacrolimus. Two months post-transplant, she was admitted for non-neutropenic fevers and diagnosed with Clostridium difficile colitis, acute kidney injury, EBV viremia (>800000 IU/ml), and widespread lymphadenopathy concerning for EBV-associated biopsy proven monomorphic PTLD. She was treated with two doses of Rituximab. Due to worsening encephalopathy, liver function tests and metabolic acidosis, ICU transfer initiated for continuous renal replacement therapy (CRRT). She developed acute respiratory failure and septic shock; was intubated, requiring vasopressors. During her ICU stay, HLH was considered. Bone marrow aspiration and biopsy performed confirmed HLH. High dose dexamethasone therapy was initiated. She also received two doses of Etoposide and a dose of tocilizumab in ICU. She was successfully extubated to high flow nasal cannula. Next Day, given her worsening mental status; Emapalumab (GAMIFANT, Novimmune SA), a fully human anti-IFN-gamma monoclonal antibody for HLH, was administered. Her cognitive function further declined, and she progressed to multiorgan failure and death.DISCUSSION: Aplastic anemia is a hematologic disorder characterized by pancytopenia and bone marrow aplasia. Bone marrow transplantation and immunosuppressive therapy are the modalities currently used, with success rates ranging 60% to 80%. Most treatment options for EBV-HLH have unclear effectiveness in adults.
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