Keywords
pedicled omentoplasty - omentum - rheumatoid pleural nodules - intractable secondary
pneumothorax - rheumatologists
Introduction
Approximately 1% of patients with rheumatoid arthritis have pleuropulmonary lesions.
Rheumatoid arthritis–related pulmonary nodules tend to necrose and rupture, leading
to the development of pneumothorax or, more severely, bronchopleural fistula.[1] Rheumatoid nodule–associated pneumothorax usually recurs and requires repeated intervention,
and is sometimes refractory to treatment.[2] This report describes a case of successful pedicle omentoplasty for difficult-to-control
rheumatoid nodule–associated pneumothorax. Routine follow-up with computed tomography
(CT) imaging revealed repeated increases and decreases in postoperative rheumatoid
nodules. However, the pneumothorax did not recur, suggesting that a strong adhesion
had been achieved.
Case Description
In 2020, a rheumatologist referred a male patient in his 50s to our thoracic department
for a first right pneumothorax caused by a rheumatoid nodule rupture ([Fig. 1]). He was being administered methotrexate and steroids to manage rheumatoid arthritis
that started 10 years earlier. No other preexisting conditions were noted. The patient
presented with pneumothorax in addition to rheumatoid arthritis symptoms and exhaustion.
Fig. 1 Chest radiograph at the time of referral to our department. The right pneumothorax
is presented here. Computed tomography image showing multiple rheumatoid nodules.
The rheumatoid nodules around the pneumothorax cavity were contiguous.
The pneumothorax cavity gradually enlarged over 2 months, and the patient underwent
thoracic drainage. Repeated pleurodesis was performed because of the presence of leaks;
however, it was ineffective. The patient expressed a preference for the treatment
to be completed in one operation as much as possible.
Spigot filling is generally used when pleurodesis fails. However, in this case, the
nodules were widely contiguous, and the feasibility of Spigot filling was uncertain;
therefore, it was not performed.
The patient underwent surgery 3 months after the referral to our department under
general anesthesia. Although a pleural defect was identified ([Fig. 2]), the surrounding area was fragile, and direct suturing was not performed because
it was judged to increase the wound size. Due to the expectation of widespread adhesion,
we performed parietal pleural covering.[3] A TachoSil covering was placed over the fistula ([Fig. 2]), and the parietal pleura was dropped over the TachoSil. The patient remained in
good condition for approximately 2 weeks, after which the pneumothorax recurred.
Fig. 2 Initial intraoperative findings are presented here. A pleural defect was observed,
and TachoSil was applied. (Red arrow)
A Clagett window and an Eloesser flap were not selected for the next course of action
because of the requirement for long-term dressing changes. Similarly, conventional
thoracoplasty was not selected because of its impact on appearance.
Although no obvious intrathoracic infection was observed at this stage, we performed
pedicled omentum filling. This choice was driven by several factors, including the
expectation of strong adhesion, the patient's immunosuppressed state, concerns regarding
future infection, and the rheumatologist's intention to use more potent antirheumatic
drugs (Janus kinase inhibitors)[4] in the future.
A second surgery was performed approximately 1 month after the initial surgery. The
pedicled omentum was harvested in a stemlike manner through a median incision in the
upper abdomen and guided into the thoracic cavity via a hole created in the diaphragm
([Fig. 3]). The patient was discharged without major adverse events postoperatively on postoperative
day 15 after pain control and rehabilitation. Regular follow-up was conducted every
month, and approximately 2 years have passed since the treatment. During the postoperative
period, the rheumatoid nodule increased and decreased repeatedly; however, we believe
that due to its firm adhesion, the pneumothorax did not recur ([Fig. 4]).
Fig. 3 Harvested pedicled omentum. Chest radiograph after filling of the pedicled omentum.
Fig. 4 Computed tomography scan of the patient after the omentum filling. Some rheumatoid
nodules are enlarged, while others are small. Despite repeated increase and decrease
in the nodules, no recurrence of pneumothorax is observed, probably because the nodules
adhered firmly (dotted line: pedicled omentum-filling area; red arrows: rheumatoid nodules over time).
Discussion
Rheumatoid arthritis presents with different symptoms in patients. Immunosuppressive
agents and other drugs used in its treatment can lead to direct pulmonary toxicity,
making patients susceptible to opportunistic infections and inhibiting wound repair
and healing.[5] Therefore, pneumothorax caused by rheumatoid nodule disruption in the lungs is likely
to be resistant to standard treatment,[2] and the treatment strategies are controversial.
In this case, the leak was initially considered to be a single point based on the
pleural findings during the initial surgery. However, CT images revealed fragility
in the surrounding area; therefore, direct suturing was deemed to be difficult. Moreover,
the rheumatoid nodules were widely contiguous, and the suitability of using Spigot
filling was uncertain. Furthermore, we applied a TachoSil + parietal pleura covering,
although it failed due to lack of firm adhesion.
The patient refused to undergo open window thoracotomy or disfigurement. Therefore,
we did not use an Eloesser flap or perform thoracoplasty. Instead, we performed pedicled
omentoplasty with the patient's consent to ensure firm adhesion of the pleura and
resistance to infection.
Pedicled omentoplasty is highly invasive because of the abdominal incision and is
not the first choice for an uninfected pulmonary fistula. However, direct suturing
is difficult for intractable pulmonary fistulas, such as that in this case, where
the surrounding lung tissue is fragile. Even if various materials are used to cover
the fistula, its closure cannot be achieved. Therefore, using pedicled omentum may
be an effective option since strong adhesion can be expected.
Rheumatoid medications were increased to control rheumatism even after filling the
omentum. However, the course of the rheumatoid nodules was mixed. No recurrence of
pneumothorax was observed approximately 2 years after omentum filling, and the patient
was satisfied with the progress. The omentum promotes angiogenesis, supplies fibroblasts,
and possesses abundant lymphatic vessels, which contribute to its strong anti-inflammatory
and self-cleansing effects. Consequently, it effectively protects against infection.
Additionally, its excellent ability to adhere to and repair adjacent tissues further
enhances its effectiveness in protecting against infection.[6]
[7]
Postoperative abdominal symptoms may occur because of the abdominal incision involved
in the omentum-filling procedure; however, in this case, none occurred.
Moreover, one problem with this treatment strategy is that it makes reopening the
chest difficult and impractical because of firm thoracic cavity adhesions. Patients
with rheumatoid arthritis are at a higher risk of pulmonary malignancy than the general
population[8]; therefore, they require careful follow-up imaging of the nodules.
In conclusion, pedicled omentoplasty is effective for rheumatoid nodule–related pneumothorax,
which differs from common pneumothorax. To the best of our knowledge, this is the
first report of a patient where pneumothorax did not recur due to firm adhesions despite
repeated increases and decreases in postoperative rheumatoid nodules, as captured
by regular CT imaging follow-ups.
