A Case of Adult-Onset Still's Disease Complicated with Diffuse Alveolar Hemorrhage

Sarı, İ̇smail; Bırlık, Merih; Binicier, Ömer Burçak; Akar, Servet; Yılmaz, Erkan; Önen, Fatoş; Akkoç, Nurullah

doi:10.3346/jkms.2009.24.1.155

Cited by 22 publications

(14 citation statements)

References 9 publications

(21 reference statements)

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“…[37,64] In addition, pulmonary hypertension may also be a thoracic complication of AOSD. In fact, pulmonary hypertension is a well-described complication of juvenile onset of Still disease.…”

Section: Discussionmentioning

confidence: 99%

Parenchymal lung involvement in adult-onset Still disease

et al. 2016

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Parenchymal lung involvement (PLI) in adult-onset Still's disease (AOSD) has seldom, if ever, been studied. We examine here retrospective cohort AOSD cases and present a review of the literature (1971–2014) on AOSD-related PLI cases.Patients with PLI were identified in 57 AOSD cases. For inclusion, the patients had to fulfill Yamaguchi or Fautrel classification criteria, show respiratory symptoms, and have imaging evidence of pulmonary involvement, and data allowing exclusion of infectious, cardiogenic, toxic, or iatrogenic cause of PLI should be available. This AOSD + PLI group was compared with a control group (non–PLI-complicated AOSD cases from the same cohort).AOSD + PLI was found in 3 out of the 57 patients with AOSD (5.3%) and the literature mentioned 27 patients. Among these 30 AOSD + PLI cases, 12 presented an acute respiratory distress syndrome (ARDS) and the remaining 18 another PLI. In the latter, a nonspecific interstitial pneumonia computed tomography pattern prevailed in the lower lobes, pulmonary function tests showed a restrictive lung function, the alveolar differential cell count was neutrophilic in half of the cases, and the histological findings were consistent with bronchiolitis and nonspecific interstitial pneumonia. Corticosteroids were fully efficient in all but 3 patients. Ten out of 12 ARDS cases occurred during the first year of the disease course. All ARDS-complicated AOSD cases received corticosteroids with favorable outcomes in 10 (2 deceased). Most PLIs occurred during the systemic onset of AOSD.PLI may occur in 5% of AOSDs, of which ARDS is the most severe. Very often, corticosteroids are efficient in controlling this complication.

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Section: Discussionmentioning

confidence: 99%

Parenchymal lung involvement in adult-onset Still disease

et al. 2016

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“…Unlike other rheumatic diseases with multiorgan involvement, in which respiratory abnormalities are well studied and documented (eg, rheumatoid arthritis, systemic lupus erythematosus, and systemic sclerosis).The pulmonary manifestations of AOSD include pleural effusion uni or bilateral (5 to 31%) or transient pulmonary infiltrates (Cheema and Quismorio 1999 ; Kozel and Sabroe 2004 ; Crispin et al 2005 ), acute and chronic pneumonitis (Kadar and Petrovicz 2004 ), functional abnormalities and miscellaneous conditions, such as diaphragmatic dysfunction, drug-induced lung disease and respiratory distress syndrome (Cheema and Quismorio 1999 ). However, to the best of our knowledge, pulmonary haemorrhage in acute presentation of AOSD has not been reported before and the previous case of Sari et al (Sari et al 2009 ), described the first case of chronic presentation of AOSD complicated with diffuse alveolar haemorrhage during the acute flare of the disease.…”

Section: Discussion and Evaluationmentioning

confidence: 93%

Acute adult-onset still’s disease presenting as pulmonary hemorrhage, urticaria, angioedema and leukemoid reaction: a case report and literature review

et al. 2015

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IntroductionAdult-onset Still’s disease is a rare systemic inflammatory disorder of unknown aetiology characterized by the classic triad of persistent high spiking fevers, joint pain and a distinctive salmon-colored bumpy rash however, the multiorgan involvement can be present.Case descriptionA 40-year-old woman previously healthy was referred to our hospital with 7 days of high fever and generalized arthralgia, The physical exam revealed angioneurotic edema detected on soles, palms and tongue and widespread red, urticated plaques in a symmetrical distribution affecting the arms, dorsal hands, upper and lower chest and back. Followed 5 days later by fever, the patient presented dyspnea, cough and hypoxemia, the imaging studies showed unilateral consolidation and pleural effusion. The bronchoscopy with bronchoalveolar lavage and skin biopsy were consistent with neutrophilic urticarial. The hematological disorders, infections and other autoimmune diseases were excluded.Discussion and evaluationThe diagnosis of adult-onset Still’s disease can be very difficult. There are no specific tests and reliance is usually placed on a symptom complex and the well described typical rash seen in most patients. In recent years, however, other cutaneous manifestations of Adult-onset Still’s disease have been reported but these are not so well known.ConclusionsThe evidence of rare manifestations is growing and the early clinical presentation of Adult-onset Still’s is extremely variable, making diagnosis difficult. For this reason, data on early clinical presentation of the disease are of interest. We reported the first case of acute Adult-onset Still’s disease with the association of pulmonary hemorrhage, urticaria and angioedema including a rare systemic manifestation as leukemoid reaction.

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“…Диффузное альвеолярное кровотечение, редкое жизнеугрожающее осложнение БСРВ, характеризуется накоплением эритроцитов в альвеолах легких и появ-лением кровохарканья, кашля и одышки, прогрессирующей до респираторного дистресса, что сопровождается падением гематокрита и геморрагической жидкостью, полученной при бронхоальвеолярном лаваже. Лечение заключается во внутривенном введении высоких доз метилпреднизолона (1 г/сут в течение 5 дней), обмене плазмы или внутривенном введении циклофосфамида [113,114].…”

Section: таблицаunclassified