ABSTRACT
Chylous effusion is a rare manifestation of systemic lupus erythematosus (SLE). When it does occur in SLE, it is generally well treated with standard pharmacologic or surgical measures. We present a decade of management in a case of SLE with lung affliction and development of refractory bilateral chylous effusion and pulmonary arterial hypertension (PAH). In the first years, the patient was treated under a Sjogren syndrome diagnose. After few years, her respiratory condition worsened due to chylous effusion and PAH. Immunosuppression therapy (methylprednisolone) was reintroduced, and vasodilator therapy commenced. With this, her cardiac function remained stable, but respiratory function continuously worsened despite several therapy trials with different combinations of immunosuppressant (glucocorticoids, resochin, cyclophosphamide and mycophenolate mofetil). On top of pleural effusion worsening, the patient developed ascites and severe hypoalbuminaemia. Even though albumin loss was stabilized with monthly octreotide applications, the patient remained respiratory insufficient and in need of continuous oxygen therapy. At that point, we decided to introduce sirolimus on top of glucocorticoids and mycophenolate mofetil therapy. Her clinical status, radiological finding, and lung function gradually improved and she became respiratory sufficient at rest. The patient remains in our follow-up and has been stable on given therapy for over 3 years despite overcoming a severe COVID-19 pneumonia in 2021. This case adds to the body of evidence of sirolimus effectiveness in patients with refractory systemic lupus and is, to our best knowledge, the first case to report its successful application in a patient with SLE and refractory chylous effusion.
Subject(s)
COVID-19 , Lupus Erythematosus, Systemic , Humans , Female , Sirolimus/therapeutic use , Glucocorticoids/therapeutic use , Mycophenolic Acid/therapeutic use , Lupus Erythematosus, Systemic/complications , Lupus Erythematosus, Systemic/diagnosis , Lupus Erythematosus, Systemic/drug therapyABSTRACT
The zoonotic potential of Mycobacterium tuberculosis complex species is well known. However, M. pinnipedii, the causative agent of tuberculosis (TB) predominantly in seals and sea lions, has never been isolated from a respiratory specimen in humans. Here we describe the first known human case of pulmonary TB caused by M. pinnipedii in a 79-year-old female patient with rheumatoid arthritis and chronic respiratory disease. The epidemiological data did not explain where the patient was exposed to M. pinnipedii, thus leaving the source of transmission unknown.
Subject(s)
Arthritis, Rheumatoid , Mycobacterium/isolation & purification , Pulmonary Disease, Chronic Obstructive , Tuberculosis, Pulmonary/diagnosis , Aged , Animals , Caniformia/microbiology , Croatia , Diagnosis, Differential , Fatal Outcome , Female , Humans , Tuberculosis, Pulmonary/diagnostic imaging , Tuberculosis, Pulmonary/microbiology , Zoonoses/diagnosis , Zoonoses/microbiologySubject(s)
Bone Neoplasms/diagnosis , Fractures, Spontaneous/diagnostic imaging , Hemangioma/diagnosis , Osteolysis/diagnostic imaging , Pleural Effusion/diagnostic imaging , Rib Fractures/diagnostic imaging , Adult , Bone Neoplasms/complications , Bone Neoplasms/pathology , Fractures, Spontaneous/etiology , Hemangioma/complications , Hemangioma/pathology , Humans , Male , Osteolysis/etiology , Pleural Effusion/etiology , Rib Fractures/etiologyABSTRACT
Lung transplant (LuTx) recipients represent a population at risk of nontuberculous mycobacterial pulmonary disease (NTM-PD). Yet the risk factors, the timing of NTM-PD after transplantation, and the association with allograft dysfunction all remain poorly defined. We report 2 cases of early-onset NTM-PD and review the literature, focusing on NTM-PD in LuTx recipients not colonized with NTM prior to transplantation. In addition, we summarize the main characteristics and differences between early- and late-onset disease.
