Pneumothorax as a first manifestation of SS

Watanabe, Satoshi; Tambo, Yuichi; Waseda, Yuko; Nishimura, Osamu; Murakami, Shinya; Tsujibata, Akihiko; Fujimura, Masaki

doi:10.1093/rheumatology/kes013

Cited by 2 publications

(4 citation statements)

References 6 publications

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“…subcutaneous emphysema) constitute very rare acute respiratory events in CTDs and may occur simultaneously or in isolation. They have been described in the literature in a variety of case reports concerning each one of the major seven CTDs, and very scarcely do they represent spontaneous events or events at initial presentation of the disease [154,155,156,157,158,159,160,161,162,163,164,165,166,167,168,169]. Most of the times, they are secondary to a preexisting chronic lung disease such as ILD with subpleural cystic-honeycombing lesions, especially in RA, scleroderma, DM-PM, and mixed connective tissue disease; the presence of peripheral necrobiotic lung nodules in RA, cystic lung disease or lower respiratory tract infections in SLE and Sjögren's syndrome, and fibrobullous disease of the lungs in ankylosing spondylitis further increases the risk of their development and aggravates their outcome.…”

Section: Pneumothorax-pneumomediastinummentioning

confidence: 99%

Acute Respiratory Events in Connective Tissue Disorders

Papiris¹,

Manali²,

Kolilekas³

et al. 2016

Respiration

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Subacute-acute, hyperacute, or even catastrophic and fulminant respiratory events occur in almost all classic connective tissue disorders (CTDs); they may share systemic life-threatening manifestations, may precipitously lead to respiratory failure requiring ventilatory support as well as a combination of specific therapeutic measures, and in most affected patients constitute the devastating end-of-life event. In CTDs, acute respiratory events may be related to any respiratory compartment including the airways, lung parenchyma, alveolar capillaries, lung vessels, pleura, and ventilatory muscles. Acute respiratory events may also precipitate disease-specific extrapulmonary organ involvement such as aspiration pneumonia and lead to digestive tract involvement and heart-related respiratory events. Finally, antirheumatic drug-related acute respiratory toxicity as well as lung infections related to the rheumatic disease and/or to immunosuppression complete the spectrum of acute respiratory events. Overall, in CTDs the lungs significantly contribute to morbidity and mortality, since they constitute a common site of disease involvement; a major site of infections related to the ‘mater' disease; a major site of drug-related toxicity, and a common site of treatment-related infectious complications. The extreme spectrum of the abovementioned events, as well as the ‘vicious' coexistence of most of the aforementioned manifestations, requires skills, specific diagnostic and therapeutic means, and most of all a multidisciplinary approach of adequately prepared and expert scientists. Avoiding lung disease might represent a major concern for future advancements in the treatment of autoimmune disorders.

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Section: Pneumothorax-pneumomediastinummentioning

confidence: 99%

Acute Respiratory Events in Connective Tissue Disorders

Papiris¹,

Manali²,

Kolilekas³

et al. 2016

Respiration

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“…Furthermore, pneumothorax was the first presentation of pSS in two cases, including ours. Both the previously documented cases had bilateral diffuse cysts on the CT chest [ 3 , 4 ]; nonetheless, our patient had only a single apical bulla reported intraoperatively, and a ruptured subpleural cyst could have caused the pneumothorax development.…”

Section: Discussionmentioning

confidence: 61%

“…To the best of our knowledge, only two cases of pneumothorax associated with pSS have been reported [ 3 , 4 ]. Table 3 illustrates the reported cases' main findings.…”

Section: Discussionmentioning

confidence: 99%

“…SS is frequently associated with numerous respiratory complications, including tracheobronchial sicca, small airway disease, various patterns of interstitial lung disease, lymphoproliferative disease, pulmonary hypertension, pleural involvement, and pulmonary amyloidosis [ 2 ]. However, the literature has published only two cases of spontaneous pneumothorax associated with SS [ 3 , 4 ]. We report a rare case of massive unilateral pneumothorax associated with SS.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Triple-Negative Sjogren’s Syndrome and Recurrent Pneumothorax: An Uncommon Presentation of Autoimmune Disease

Imad¹,

Abdalla²,

Hamza³

et al. 2022

Cureus

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Sjogren's syndrome (SS) is a chronic exocrinopathy caused by lymphocytic infiltration and is associated with numerous manifestations and morbidities. We discuss a case of a 60-year-old female who presented to the Acute Medical Assessment Unit complaining of progressive shortness of breath for one month, not associated with chest pain or lower limb swelling. She also reported joint pain involving both wrists and proximal interphalangeal (PIP) joints, oral dryness, hair loss, and numerous tongue ulcerations. Blood workup revealed triple-negative SS, negative rheumatoid factor, anti-SSA and anti-SSB, a high erythrocyte sedimentation rate (ESR), and antinuclear antibody (ANA) titer of 640. A diagnosis of SS was made. Nevertheless, her CT chest showed massive left-sided pneumothorax; subsequently, a chest tube was urgently inserted. The chest tube was removed two days later with complete resolution on chest X-ray (CXR). However, one week later, she presented with a recurrent pneumothorax that persisted and required surgical intervention that led to complete recovery afterward. Pneumothorax is an extremely rare but potentially unfavorable complication related to SS, with only two cases reported in the literature so far and usually associated with underlying lung pathology.

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Pneumothorax as a first manifestation of SS

Cited by 2 publications

References 6 publications

Acute Respiratory Events in Connective Tissue Disorders

Acute Respiratory Events in Connective Tissue Disorders

Triple-Negative Sjogren’s Syndrome and Recurrent Pneumothorax: An Uncommon Presentation of Autoimmune Disease

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