Skip to main content

Advertisement

SpringerLink
Log in

Pneumomediastinum, pneumatosis intestinalis and pneumoperitoneum in a patient with polymyositis: case-based review

  • Case Based Review
  • Published:
Rheumatology International Aims and scope Submit manuscript

Abstract

Pneumomediastinum (PnM), pneumatosis intestinalis (PI), and pneumoperitoneum (PP) are rare complications of inflammatory myositis. We present a 59-year-old polymyositis (PM) patient who experienced all three complications simultaneously. The patient who presented with proximal muscle weakness, dysphagia, and weight loss was diagnosed with PM due to elevated muscle enzymes and consistent electromyography and muscle biopsy with inflammatory myopathy. On the 45th day of her immunosuppressive treatment, PnM, PI, and PP were detected incidentally in 18F-fluorodeoxyglucose (FDG) positron emission tomography (PET)/computed tomography (CT) scan performed for severe weight loss and treatment-resistant severe disease. Since the patient had no symptoms or signs of PnM and PP, no additional intervention was applied to the current treatment, and spontaneous regression was observed in the follow-up. In addition to this case, we reviewed patients with PM who developed PBM, PP, and PI in the literature.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. Kouritas VK, Papagiannopoulos K, Lazaridis G, Baka S, Mpoukovinas I, Karavasilis V et al (2015) Pneumomediastinum 7:44–49. https://doi.org/10.3978/j.issn.2072-1439.2015.01.11

    Article  Google Scholar 

  2. Mularski RA, Sippel JM, Osborne ML (2000) Pneumoperitoneum: a review of nonsurgical causes. Crit Care Med 28:2638–2644. https://doi.org/10.1097/00003246-200007000-00078

    Article  CAS  PubMed  Google Scholar 

  3. Saito M, Tanikawa A, Nakasute K, Tanaka M, Nishikawa T (2007) Additive contribution of multiple factors in the development of pneumatosis intestinalis: a case report and review of the literature. Clin Rheumatol 26:601–603. https://doi.org/10.1007/s10067-005-0179-9

    Article  PubMed  Google Scholar 

  4. Le Goff B, Chérin P, Cantagrel A, Gayraud M, Hachulla E, Laborde F et al (2009) Pneumomediastinum in interstitial lung disease associated with dermatomyositis and polymyositis. Arthritis Rheum 61:108–118. https://doi.org/10.1002/art.24372

    Article  PubMed  Google Scholar 

  5. Zhang L, Shen M, Zhang F, Tang F (2014) Survival analysis and risk factors for mortality in connective tissue disease-associated pneumomediastinum. Rheumatol Int 34:1657–1663. https://doi.org/10.1007/s00296-014-3046-7

    Article  CAS  PubMed  Google Scholar 

  6. Zarbalian Y, von Rosenvinge EC, Twadell W, Mikdashi J (2013) Recurrent pneumatosis intestinalis in a patient with dermatomyositis. BMJ Case Rep 2013:bcr2013200308. https://doi.org/10.1136/bcr-2013-200308

    Article  PubMed  PubMed Central  Google Scholar 

  7. Kim JJ, Kim D, Kim EK, Sohn IW, Jung KH, Choi C-B et al (2011) Clinical significance of spontaneous pneumomediastinum in dermatomyositis/polymyositis. Korean J Gastroenterol 57:249–252. https://doi.org/10.4166/kjg.2011.57.4.249

    Article  Google Scholar 

  8. Maruoka H, Honda S, Takeo M, Kitazato H, Hanai N, Ayukawa R et al (2006) A case of polymyositis complicated with interstitial pneumonitis and pneumomediastinum. Mod Rheumatol 16:55–57. https://doi.org/10.1007/s10165-005-0451-5

    Article  PubMed  Google Scholar 

  9. Shimamoto K, Ozaki Y, Amuro H, Son Y, Ota-Imamura M, Tanijiri T et al (2008) Three cases of polymyositis/dermatomyositis complicated by pneumomediastinum. Nihon Rinsho Meneki Gakkai Kaishi 31:56–61. https://doi.org/10.2177/jsci.31.56

    Article  PubMed  Google Scholar 

  10. Okada R, Miyabe YS, Kasai S, Hashimoto K, Yamauchi S, Yoshikawa M et al (2010) Successful treatment of interstitial pneumonia and pneumomediastinum associated with polymyositis during pregnancy with a combination of cyclophosphamide and tacrolimus: a case report. Nihon Rinsho Meneki Gakkai Kaishi 33:142–148. https://doi.org/10.2177/jsci.33.142

    Article  PubMed  Google Scholar 

  11. Wada Y, Murayama N, Hirose S, In H, Kuroda T, Ito S et al (2004) A case of pneumatosis cystoides intestinalis in a patient with polymyositis and interstitial pneumonia. Mod Rheumatol 14:260–263. https://doi.org/10.1007/s10165-004-0304-7

    Article  PubMed  Google Scholar 

  12. Hanawa M, Toda H, Kobayashi H et al (1982) Interstitial pneumonia, intestinal pseudo-obstruction and pneumatosis cystoides intestinalis associated with polymyositis: report of a case. Stomach Intestine 2013:1021–1027

    Google Scholar 

  13. Elkayam O, Caspi D, Flusser G (2001) Pneumatosis intestinalis in a patient with polymyositis. Clin Exp Rheumatol 19:483

    CAS  PubMed  Google Scholar 

  14. Park YH, Kanoh T, Nishida O, Yoshida Y, Ohkuma M, Uchino H et al (1985) A case of polymyositis with ileus-like symptoms and pneumatosis cystoides intestinalis, improved by the treatment of high-flow oxygen. Nippon Naika Gakkai Zasshi 74:808–812. https://doi.org/10.2169/naika.74.808

    Article  CAS  PubMed  Google Scholar 

  15. Kuroda T, Ohfuchi Y, Hirose S, Nakano M, Gejyo F, Arakawa M (2001) Pneumatosis cystoides intestinalis in a patient with polymyositis. Clin Rheumatol 20:49–52. https://doi.org/10.1007/pl00011184

    Article  CAS  PubMed  Google Scholar 

  16. Kim JY, Kwon YH, Lee SJ, Jang SY, Park SY (2011) Pneumatosis cystoides ıntestinalis and partial abdominal obstruction in a patient with polymyositis. Intest Res 9:234–237. https://doi.org/10.5217/ir.2011.9.3.234

    Article  Google Scholar 

  17. Lee SJ, Park JY, Kwon SA, Koh DH, Choi MH, Jang HJ et al (2011) A case of pneumatosis cystoids intestinalis with polymyositis. Korean J Gastroenterol 57:249–252. https://doi.org/10.4166/kjg.2011.57.4.249

    Article  PubMed  Google Scholar 

  18. Nakano H, Shibata T, Yoshida A, Maeda A, Takakuwa Y, Oka H et al (2008) A case of polymyositis complicated with pneumatosis intestinalis and pneumomediastinum. Clin Rheumatol 20:41–46. https://doi.org/10.14961/cra.20.41

    Article  Google Scholar 

  19. De Giacomi F, Baqir M, Cox CW, Moua T, Matteson EL, Ryu JH (2019) Spontaneous pneumomediastinum in connective tissue diseases. J Clin Rheumatol 25:239–245. https://doi.org/10.1097/RHU.0000000000000835

    Article  PubMed  Google Scholar 

  20. Yamanishi Y, Maeda H, Konishi F, Hiyama K, Yamana S, Ishioka S et al (1999) Dermatomyositis associated with rapidly progressive fatal interstitial pneumonitis and pneumomediastinum. Scand J Rheumatol 28:58–61. https://doi.org/10.1080/03009749950155805

    Article  CAS  PubMed  Google Scholar 

  21. Kono H, Inokuma S, Nakayama H, Suzuki M (2000) Pneumomediastinum in dermatomyositis: association with cutaneous vasculopathy. Ann Rheum Dis 59:372–376. https://doi.org/10.1136/ard.59.5.372

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  22. Than VS, Nguyen MD, Gallon A, Pham MT, Nguyen DH, Boyer L et al (2020) Pneumatosis intestinalis with pneumoperitoneum: not always a surgical emergency. Radiol Case Rep 15:2459–2463. https://doi.org/10.1016/j.radcr.2020.09.034

    Article  PubMed  PubMed Central  Google Scholar 

Download references

Funding

None.

Author information

Authors and Affiliations

  1. Division of Rheumatology, Department of Internal Medicine, Faculty of Medicine, Akdeniz University, 07070, Antalya, Turkey

    Melis Dilbil, Funda Erbasan, Bengisu Aslan, Tahir Saygın Öğüt, Mine Nokay, Veli Yazısız & Mustafa Ender Terzioğlu

Authors
  1. Melis Dilbil
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Funda Erbasan
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Bengisu Aslan
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Tahir Saygın Öğüt
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Mine Nokay
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Veli Yazısız
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Mustafa Ender Terzioğlu
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

MD, TSÖ, and MN reviewed the literature, MD and FE analyzed the cases and wrote the manuscript, and VY and MET approved the final version of the manuscript.

Corresponding author

Correspondence to Funda Erbasan.

Ethics declarations

Conflict of interest

The authors declare that there is no conflict of interest.

Ethical approval

This study was conducted in compliance with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from the patient for publication.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Springer Nature or its licensor holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Dilbil, M., Erbasan, F., Aslan, B. et al. Pneumomediastinum, pneumatosis intestinalis and pneumoperitoneum in a patient with polymyositis: case-based review. Rheumatol Int 43, 771–776 (2023). https://doi.org/10.1007/s00296-022-05205-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00296-022-05205-1

Keywords

Search

Navigation