CASE REPORT Open Access

Intractable pneumothorax due to ruptureof subpleural rheumatoid nodules: a casereportMasanori Shimomura* , Shunta Ishihara and Masashi Iwasaki

Abstract

Background: In rare cases, rheumatoid pleural nodules can rupture into the pleural cavity to causepneumothorax or empyema. We report successful surgical treatment of a patient with an intractablesecondary pneumothorax due to rupture of a subpleural rheumatoid nodule into the pleural cavity.

Case presentation: A 75-year-old man with a medical history of rheumatoid arthritis, acute coronarysyndrome, and diabetes was admitted to our hospital because of left chest pain and dyspnea. A chest X-rayand chest computed tomography (CT) scan showed a left pneumothorax and several small subpleuralnodules with cavitation. Repeated pleurodesis via a chest tube failed to improve the pneumothorax, so wedecided to perform thoracoscopic surgery. Air leakage was detected in the left upper lobe where thesubpleural nodule was visible on chest CT. Resection of the lesion successfully resulted in resolution of the airleakage. The final pathological diagnosis of the subpleural nodule was a pulmonary rheumatoid nodule. Thepatient has had no evidence of recurrence of pneumothorax after surgery.

Conclusions: We obtained a final pathological diagnosis of a rheumatoid nodule that caused an intractablepneumothorax. Pneumothorax associated with rupture of rheumatoid nodules in the subpleural cavitary isdifficult to treat with thoracoscopic surgery as a second-line treatment.

Keywords: Secondary pneumothorax, Rheumatoid arthritis, Thoracoscopic surgery, Pleurodesis

BackgroundRheumatoid arthritis (RA) is the most common formof chronic inflammatory arthritis; RA can causenoncardiac thoracic complications, such as pleuritis,emphysema, pneumothorax, interstitial pneumonia,and rheumatoid nodules [1]. Noncardiac thoraciccomplications of RA occur in 5–20% of RA patientsand account for approximately 20% of RA-associatedmortality [2–4]. We report successful surgical treat-ment of an intractable secondary pneumothorax dueto rupture of a subpleural rheumatoid nodule into thepleural cavity.

Case presentationA 75-year-old man was admitted to our hospital be-cause of left chest pain and dyspnea. A chest X-rayshowed a left pneumothorax (Fig. 1), and a previouschest computed tomography (CT) scan revealed asubpleural pulmonary nodule with cavitation in theleft upper lobe (Fig. 2). The patient had been onprednisolone and methotrexate for rheumatoid arth-ritis. He also had a past history of acute coronarysyndrome 5 years previously; this was treated withcoronary stent implantation followed by anticoagulanttherapy. He additionally had a past history of a rightpneumothorax 3 years previously that was presump-tively caused by rupture of a subpleural rheumatoidnodule and successfully treated with conservativethoracic drainage. Regarding his present admission,the left pneumothorax did not resolve after 1 week of

* Correspondence: shimomu@hera.eonet.ne.jpDepartment of General Thoracic Surgery, Ayabe City Hospital, 20-1 Otsuka,Aono-cho, Ayabe, Kyoto 623-0011, Japan

© The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made.

Shimomura et al. Surgical Case Reports (2018) 4:89 https://doi.org/10.1186/s40792-018-0502-8

chest tube drainage. There were no bacterial patho-gens present in the pleural effusion. We decided toperform pleurodesis because his left lung expandedwell after chest drainage and he had some risks forpneumothorax surgery, as previously mentioned. Theair leakage finally subsided after six times pleurodesisprocedures (with a blood patch once, a talc slurrythree times, and OK-432 (streptococcal preparation)two times) prior to discharge (Fig. 1b). However, hepresented to our hospital 1 week after being dis-charged because the pneumothorax had asymptomati-cally relapsed (Fig. 1c). Thus, we decided to performthoracoscopic surgery to resect the subpleural lesionthat caused intractable pneumothorax 2 months afterthe first admission. The surgery was performedthrough the fourth intercostal space along the mid-axillary line, sixth intercostal space along the ante-roaxillary line, and seventh intercostal space along

the posteroaxillary line. There were rigid adhesionson the apex, mediastinum, and diaphragm due to re-petitive pleurodesis. A sealing test revealed a pleuralfistula with air leakage of the left upper lobe wherethe previous chest CT scan had demonstrated asmall subpleural nodule with cavitation (Fig. 3a).The visceral pleura was so thick and hard that wehad to resect the lesion with surgical scissors insteadof with automatic staplers. The resected lung paren-chyma was continuously sutured with 4-0 Prolene®(Ethicon endo-surgery) and covered with a polygly-colic acid sheet (Fig. 3b). There was no air leakageat the end of surgery. The lung gradually expanded(Fig. 4) for a few days. The chest tube was success-fully removed on the third postoperative day, andthe patient was discharged on the seventh postopera-tive day. The final pathological diagnosis was a pul-monary rheumatoid nodule on the pleura (Fig. 5).The patient has had no evidence of recurrence ofpneumothorax during 1 year of follow-up.

DiscussionPulmonary rheumatoid nodules occur in less than1% of patients with RA [5]. Rheumatoid nodules canrupture into the pleural cavity and cause pneumo-thorax, empyema, and bronchopleural fistula (BPF).Some reports also indicate that rapid growth ofrheumatoid nodules may be associated withazathioprine treatment [6] or biological therapies [7]of RA. Kim et al. also reported a case of recurrentpneumothorax after etanercept therapy induced bythe progression of interstitial lung disease [8].Pneumothorax caused by noncardiac intrathoraciccomplications of RA can be resistant to standardcare because RA patients are immunosuppressed,which predisposes them to infection and poor heal-ing. Some authors have reported difficulties in

Fig. 1 Chest radiograph showing a left pneumothorax on admission (a). The lung expansion improved after repeat pleurodesis (b). A relapsedleft pneumothorax occurred on readmission (c)

Fig. 2 Chest computed tomography before pneumothorax showinga subpleural nodule with cavitation (arrow)

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performing surgical intervention for pneumothoraxinduced by RA. Rueth et al. reported that some pa-tients with RA-induced pneumothorax and BPF re-quired long-term care and had pneumothoraces thatwere resistant to repeat talc pleurodesis [9]. The suc-cess rate for pleurodesis via thoracoscopic surgeryfor the management of pneumothorax in patientswithout RA is 97%, and the long-term recurrencerates range from 5 to 9% [10]. Regarding pneumo-thorax in patients with RA, we hypothesize that thecontrol rate for pneumothorax is lower than in thosewithout RA because RA patients have poor wound

healing and chronic local inflammation in the causa-tive lesions. Due to the intrathoracic rigid adhesionsthat form surrounding the causative fistula after re-petitive pleurodesis, pleurodesis can be useful fortreating patients with multiple rheumatoid nodulesbecause this procedure can prevent recurrentpneumothorax resulting from subsequent rupture ofother nodules. On the other hand, surgery to treatany residual air leakage is very difficult because ofadhesions that have formed at other locations notcontributing to the air leakage and due to the thickvisceral pleura. A wide pleural covering techniquewith an oxidized regenerated cellulose mesh [11] ora polyglycolic acid sheet can be used to treat intract-able pneumothorax rather than fistula resection andsuturing in patients in whom the cause of pneumo-thorax are still unknown at the time of surgery. Theuse of a latissimus dorsi muscle flap placed into thepleural cavity can also be effective because this flapis made with a large muscle with a reliable bloodsupply and is relatively easy to harvest. Bronchial oc-clusion with a spigot is also one of the most effect-ive techniques for patients with poor conditions forsurgery [12]. In our patient, the causative lesion wassuspected to be solitary, and it was difficult to treatthe air leakage with bronchial occlusion because thelesion was located in the peripheral lung paren-chyma. Additionally, we selected surgical resectionto pathologically demonstrate that the definitive eti-ology of the pneumothorax was rupture of arheumatoid nodule. In RA patients, it is importantto obtain a pathological diagnosis of the underlyingcause of pneumothorax because these patients havehigher risks of lung malignancies than the generalpopulation [13].

Fig. 3 Intraoperative findings of a fistula from the rheumatoid nodule (a). The lung parenchyma was sutured continuously after resection of therheumatoid nodule (b)

Fig. 4 Postoperative chest radiograph showing full expansion of the lung

Shimomura et al. Surgical Case Reports (2018) 4:89 Page 3 of 5

ConclusionsWe obtained a final pathological diagnosis of arheumatoid nodule that caused an intractablepneumothorax. Pneumothorax associated with ruptureof rheumatoid nodules in the subpleural cavitary isdifficult to treat with thoracoscopic surgery as asecond-line treatment.

AbbreviationsBPF: Bronchopleural fistula; CT: Computed tomography; RA: Rheumatoidarthritis

Availability of data and materialsThe dataset supporting the conclusion of this article is included within thearticle.

Authors’ contributionsAll authors equally participated in the care of the patient. All authorsparticipated in the acquisition, analysis, or interpretation of the data;drafting and revising of the manuscript; final approval of the paper; andagreement to be accountable for the integrity of the case report. Allauthors read and approved the final manuscript.

Ethics approval and consent to participateNot applicable

Consent for publicationThe patient provided permission to publish the features of his case. Theidentity of this patient has been protected.

Competing interestsThe authors declare that they have no competing interests.

Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.

Received: 11 June 2018 Accepted: 2 August 2018

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Fig. 5 Pathological diagnosis of the resected specimen. A subpleural rheumatoid nodule with cavitation and a defect in the pleura where thecavity had formed (a loupe image). A highly magnified view of the lesion showing fibrinoid necrosis surrounded by histiocytes and lymphocytes(b HE, × 40; c HE, × 100)

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