Skip to main content
Top
Published in: BMC Gastroenterology 1/2019

Open Access 01-12-2019 | Computed Tomography | Case report

Pneumatosis cystoides intestinalis: a case report and literature review

Authors: Fangmei Ling, Di Guo, Liangru Zhu

Published in: BMC Gastroenterology | Issue 1/2019

Login to get access

Abstract

Background

Pneumatosis cystoides intestinalis (PCI) is a low-incidence disease that confuses many doctors. A vast number of factors are suspected to contribute to its pathogenesis, such as Crohn’s disease, intestinal stenosis, ulcerative colitis, drug use, extra-gastrointestinal diseases, and chronic obstructive pulmonary disease. Most consider its pathogenesis interrelated to an increase in intra-intestinal pressure and the accumulation of gas produced by aerogenic bacteria, and patients with atypical symptoms and imaging manifestations tend to be misdiagnosed.

Case presentation

A 64-year-old man complained of a 3-month history of bloody stool without mucopurulent discharge, abdominal pain, or diarrhea. Colonoscopy revealed multiple nodular projections into the segmental mucosa of the sigmoid colon. Crohn’s disease and malignant disease ware suspected first according to the patient’s history, but laboratory examinations did not confirm either. Endoscopic ultrasound (EUS) revealed multiple cystic lesions in the submucosa. Moreover, computer tomography scan showed multiple bubble-like cysts. Combined with ultrasonography, computed tomography, and pathology findings, we ultimately made a diagnosis of PCI. Instead of surgery, we recommended conservative treatment consisting of endoscopy and oral drug administration. His symptoms improved with drug therapy after discharge, and no recurrence was noted on follow-up.

Conclusions

The incidence of PCI is low. Due to a lack of specificity in clinical manifestations and endoscopic findings, it often misdiagnosed as intestinal polyps, tumors, inflammatory bowel disease, or other conditions. Colonoscopy, computed tomography, and ultrasonography have demonstrated benefit in patients with multiple nodular projections in colon. Compared to the treatment of the above diseases, PCI treatment is effective and convenient, and the prognosis is optimistic. Therefore, clinicians should increase their awareness of PCI to avoid unnecessary misdiagnosis.
Literature
1.
go back to reference Khail PN, Huber-Wagner S, Ladurner R, Kleespies A, Siebeck M, Mutschler W, Halfeldt K, Kanz KG. Natural history, clinical pattern, and surgical consideration of pneumatosis interstialis. Eur J Med Res. 2009;14(6):231–9.CrossRef Khail PN, Huber-Wagner S, Ladurner R, Kleespies A, Siebeck M, Mutschler W, Halfeldt K, Kanz KG. Natural history, clinical pattern, and surgical consideration of pneumatosis interstialis. Eur J Med Res. 2009;14(6):231–9.CrossRef
2.
3.
go back to reference Boerner RM, Frie DB, Warshauer DM, Isaacs K. Pneumatosis intestinalis. Two case reports and a retrospective review of literature from 1985 to 1995. Dig Dis Sci. 1996;41(11):2272–85.PubMedCrossRef Boerner RM, Frie DB, Warshauer DM, Isaacs K. Pneumatosis intestinalis. Two case reports and a retrospective review of literature from 1985 to 1995. Dig Dis Sci. 1996;41(11):2272–85.PubMedCrossRef
4.
go back to reference Greenstein AJ, Nguyen SQ, Berlin A, Corona J, Lee J, Wong E, Factor SH, Divino CM. Pneumatosis intestinalis in adults: managements, surgical indivations, and risk factors for mortality. J Gastrointest Surg. 2007;11(10):1268–74.PubMedCrossRef Greenstein AJ, Nguyen SQ, Berlin A, Corona J, Lee J, Wong E, Factor SH, Divino CM. Pneumatosis intestinalis in adults: managements, surgical indivations, and risk factors for mortality. J Gastrointest Surg. 2007;11(10):1268–74.PubMedCrossRef
5.
go back to reference Keyting WS, McCarver RR, Kovarik JL, Daywitt AL. Pneumatosis intestinalis: a new concept. Radiology. 1961;76:733–41.PubMedCrossRef Keyting WS, McCarver RR, Kovarik JL, Daywitt AL. Pneumatosis intestinalis: a new concept. Radiology. 1961;76:733–41.PubMedCrossRef
7.
go back to reference Tsujimoto T, Shioyama E, Moriya K. Pneumatosis cystoides intestinalis following alpha-glucosidase inhibitor treatment: a case report and review of the literature. World J Gastroenterol. 2008;14(39):6087–92.PubMedPubMedCentralCrossRef Tsujimoto T, Shioyama E, Moriya K. Pneumatosis cystoides intestinalis following alpha-glucosidase inhibitor treatment: a case report and review of the literature. World J Gastroenterol. 2008;14(39):6087–92.PubMedPubMedCentralCrossRef
8.
go back to reference Hayakawa T, Yoneshima M, Abe T, Nomura G. Pneumatosis cystoides intestinalis after treatment with an alpha-glucosidase inhibitor. Diabetes Care. 1999;22:366–7.PubMedCrossRef Hayakawa T, Yoneshima M, Abe T, Nomura G. Pneumatosis cystoides intestinalis after treatment with an alpha-glucosidase inhibitor. Diabetes Care. 1999;22:366–7.PubMedCrossRef
9.
go back to reference Kojima K, Tsujimoto T, Fujii H, Morimoto T, Yoshioka S, Kato S, Yasuhara Y, Aizawa S, Sawai M, Makutani S, Yamamoto K, Mochi T, Fukui H. Pneumatosis cystoides intestinalis induced by the α-glucosidase inhibitor miglitol. Intern Med. 2010;49(15):1545–8.PubMedCrossRef Kojima K, Tsujimoto T, Fujii H, Morimoto T, Yoshioka S, Kato S, Yasuhara Y, Aizawa S, Sawai M, Makutani S, Yamamoto K, Mochi T, Fukui H. Pneumatosis cystoides intestinalis induced by the α-glucosidase inhibitor miglitol. Intern Med. 2010;49(15):1545–8.PubMedCrossRef
10.
go back to reference Morris MS, Gee AC, Cho SD, Limbaugh K, Underwood S, Ham B, Schreiber MA. Management and outcome of pneumatosis intestinalis. Am J Surg. 2008;195(5):679–82.PubMedCrossRef Morris MS, Gee AC, Cho SD, Limbaugh K, Underwood S, Ham B, Schreiber MA. Management and outcome of pneumatosis intestinalis. Am J Surg. 2008;195(5):679–82.PubMedCrossRef
11.
go back to reference Kanchela D, Vattikuti S, Vipperla K. Pnematosis cystoides intestinalis: is surgery always indicated? Cleve Clin J Med. 2015;82(3):151–2.CrossRef Kanchela D, Vattikuti S, Vipperla K. Pnematosis cystoides intestinalis: is surgery always indicated? Cleve Clin J Med. 2015;82(3):151–2.CrossRef
12.
go back to reference Ogul H, Pirimoglu B, Kisaoglu A, Karaca L, Havan N, Ozogul B, Kantarci M. Pneumatosis cystoides intestinalis: an unusual cause of intestinal ischemia and pneumoperitoneum. Int Surg. 2015;100(2):221–4.PubMedPubMedCentralCrossRef Ogul H, Pirimoglu B, Kisaoglu A, Karaca L, Havan N, Ozogul B, Kantarci M. Pneumatosis cystoides intestinalis: an unusual cause of intestinal ischemia and pneumoperitoneum. Int Surg. 2015;100(2):221–4.PubMedPubMedCentralCrossRef
13.
go back to reference DuBose JJ, Lissauer M, Maung AA, Piper GL, O'Callaghan TA, Luo-Owen X, Inaba K, Okoye O, Shestopalov A, Fielder WD, Ferrada P, Wilson A, Channel J, Moore FO, Paul DB, Johnson S. Pneumatosis intestinalis predictive evaluation study (PIPES):a multicenter epidemiologic study of the eastern association for the surgery of trauma. J Trauma Acute Care Surg. 2013;75(1):15–23.PubMedCrossRef DuBose JJ, Lissauer M, Maung AA, Piper GL, O'Callaghan TA, Luo-Owen X, Inaba K, Okoye O, Shestopalov A, Fielder WD, Ferrada P, Wilson A, Channel J, Moore FO, Paul DB, Johnson S. Pneumatosis intestinalis predictive evaluation study (PIPES):a multicenter epidemiologic study of the eastern association for the surgery of trauma. J Trauma Acute Care Surg. 2013;75(1):15–23.PubMedCrossRef
14.
go back to reference Ponz de Leon M, Bertarelli C, Casadei GP, Grilli A, Bacchini P, Pedroni M, Jovine E. A case of pneumatosis cystoides intestinalis mimicking familial adenomatous polyposis. Familial Cancer. 2013;12(3):573–6.PubMedCrossRef Ponz de Leon M, Bertarelli C, Casadei GP, Grilli A, Bacchini P, Pedroni M, Jovine E. A case of pneumatosis cystoides intestinalis mimicking familial adenomatous polyposis. Familial Cancer. 2013;12(3):573–6.PubMedCrossRef
15.
go back to reference Liu T, Zhang S, Mao H. Gastrointestinal malignant neoplasms disguised as pneumatosis cystoids intestinalis: a case report and literature review. Medicine (Baltimore). 2017;96(51):e9410.CrossRef Liu T, Zhang S, Mao H. Gastrointestinal malignant neoplasms disguised as pneumatosis cystoids intestinalis: a case report and literature review. Medicine (Baltimore). 2017;96(51):e9410.CrossRef
16.
go back to reference Suarez V, Chesner IM, Price AB, Newman J. Pneumatosis cystoides intestinalis. Histological mucosal changes mimicking inflammatory bowel disease. Arch Pathol Lab Med. 1989;113(8):898–901.PubMed Suarez V, Chesner IM, Price AB, Newman J. Pneumatosis cystoides intestinalis. Histological mucosal changes mimicking inflammatory bowel disease. Arch Pathol Lab Med. 1989;113(8):898–901.PubMed
17.
go back to reference Ang CH, Li XF, Ong LY, Low Y. Pneumatosis Intestinalis in meconium inspissation mimicking Necrotising Enterocolitis. Indian J Pediatr. 2017;84(4):328–9.PubMedCrossRef Ang CH, Li XF, Ong LY, Low Y. Pneumatosis Intestinalis in meconium inspissation mimicking Necrotising Enterocolitis. Indian J Pediatr. 2017;84(4):328–9.PubMedCrossRef
18.
go back to reference Johansson K, Lindström E. Treatment of obstructive pneumatosis coli with endoscopic sclerotherapy: report of a case. Dis Colon Rectum. 1991;34(1):94–6.PubMedCrossRef Johansson K, Lindström E. Treatment of obstructive pneumatosis coli with endoscopic sclerotherapy: report of a case. Dis Colon Rectum. 1991;34(1):94–6.PubMedCrossRef
19.
go back to reference Dhadie S, Mehanna D, McCourtney J. Pneumatosis intestinalis a trap for the unway: case series and literature review. Int J Surg Case Rep. 2018;53:214–7.CrossRef Dhadie S, Mehanna D, McCourtney J. Pneumatosis intestinalis a trap for the unway: case series and literature review. Int J Surg Case Rep. 2018;53:214–7.CrossRef
20.
go back to reference Lee YS, Han JJ, Kim SY, Maeng CH. Pneumatosis cystoides intestinalis associated with sunitinib and a literature review. BMC Cancer. 2017;17(1):732.PubMedPubMedCentralCrossRef Lee YS, Han JJ, Kim SY, Maeng CH. Pneumatosis cystoides intestinalis associated with sunitinib and a literature review. BMC Cancer. 2017;17(1):732.PubMedPubMedCentralCrossRef
21.
go back to reference Lassandro F, Mangoni de Santo Stefano ML, Porto AM, Grassi R, Scaglione M, Rotondo A. Intestinal pneumatosis in adults: diagnostic and prognostic value. Emerg Radiol. 2010;17(5):361–5.PubMedCrossRef Lassandro F, Mangoni de Santo Stefano ML, Porto AM, Grassi R, Scaglione M, Rotondo A. Intestinal pneumatosis in adults: diagnostic and prognostic value. Emerg Radiol. 2010;17(5):361–5.PubMedCrossRef
22.
go back to reference Ribaldone DG, Bruno M, Gaia S, Saracco GM, De Angelis C. Endoscopic ultrasound to diagnose pneumatosis cystoides intestinalis (with video). Endosc Ultrasound. 2017;6(6):416–7.PubMedPubMedCentralCrossRef Ribaldone DG, Bruno M, Gaia S, Saracco GM, De Angelis C. Endoscopic ultrasound to diagnose pneumatosis cystoides intestinalis (with video). Endosc Ultrasound. 2017;6(6):416–7.PubMedPubMedCentralCrossRef
23.
go back to reference Kaplan GG. The global burden of IBD: from 2015 to 2025. Nat Rev Gastroenterol Hepatol. 2015;12(12):720–7.PubMedCrossRef Kaplan GG. The global burden of IBD: from 2015 to 2025. Nat Rev Gastroenterol Hepatol. 2015;12(12):720–7.PubMedCrossRef
24.
go back to reference Mármol I, Sánchez-de-Diego C, Pradilla Dieste A, Cerrada E, Rodriguez Yoldi MJ. Colorectal carcinoma: a general overview and future perspective in colorectal cancer. Int J Mol Sci. 2017;18(1):197.PubMedCentralCrossRef Mármol I, Sánchez-de-Diego C, Pradilla Dieste A, Cerrada E, Rodriguez Yoldi MJ. Colorectal carcinoma: a general overview and future perspective in colorectal cancer. Int J Mol Sci. 2017;18(1):197.PubMedCentralCrossRef
25.
go back to reference Galandiuk S, Fazio VW, Petras RE. Pneumatosis cystoides intestinalis in Crohn’s disease. Report of two cases. Dis Colon Rectum. 1985;28(12):951–6.PubMedCrossRef Galandiuk S, Fazio VW, Petras RE. Pneumatosis cystoides intestinalis in Crohn’s disease. Report of two cases. Dis Colon Rectum. 1985;28(12):951–6.PubMedCrossRef
26.
go back to reference Breitinger A, Kozarek R, Hauptman E. Pneumatosis cystoides intestinalis in Crohn’s disease. Gastrointest Endosc. 2003;57(2):241.PubMedCrossRef Breitinger A, Kozarek R, Hauptman E. Pneumatosis cystoides intestinalis in Crohn’s disease. Gastrointest Endosc. 2003;57(2):241.PubMedCrossRef
27.
go back to reference Koreishi A, Lauwers GY, Misdraji J. Penmatosis intestinalis:a challenging biopsy diagnosis. Am J Surg Pathol. 2007;31(10):1469–75.PubMedCrossRef Koreishi A, Lauwers GY, Misdraji J. Penmatosis intestinalis:a challenging biopsy diagnosis. Am J Surg Pathol. 2007;31(10):1469–75.PubMedCrossRef
28.
go back to reference Beetz O, Kleine M, Vondran FWR, Cammann S, Klempnauer J, Kettler B. A case of recurrent pneumoperitoneum and pneumatosis intestinalis after bilateral lung transplan. Exp Clin Transplant. 2019;17(1):124–7.PubMedCrossRef Beetz O, Kleine M, Vondran FWR, Cammann S, Klempnauer J, Kettler B. A case of recurrent pneumoperitoneum and pneumatosis intestinalis after bilateral lung transplan. Exp Clin Transplant. 2019;17(1):124–7.PubMedCrossRef
29.
go back to reference Day DL, Ramsay NK, Letourneau JG. Pneumatosis intestinalis after bone marrow transplantation. AJR Am J Roentgenol. 1988;151(1):85–7.PubMedCrossRef Day DL, Ramsay NK, Letourneau JG. Pneumatosis intestinalis after bone marrow transplantation. AJR Am J Roentgenol. 1988;151(1):85–7.PubMedCrossRef
30.
go back to reference Marinello DK, Rafael D, Paiva Edos S, Dominoni RL. Systemic lupus erythematosus complicated by intestinal vasculitis and pneumatosis intestinalis. Rev Bras Reumatol. 2010;50:596–602.PubMedCrossRef Marinello DK, Rafael D, Paiva Edos S, Dominoni RL. Systemic lupus erythematosus complicated by intestinal vasculitis and pneumatosis intestinalis. Rev Bras Reumatol. 2010;50:596–602.PubMedCrossRef
31.
32.
go back to reference Nakatani K, Kato T, Okada S, Matsumoto R, Nishida K, Komuro H, Suganuma T. Successful treatment with hyperbaric oxygen therapy for pneumatosis cystoides intestinalis as a complication of granulomatosis with polyangiitis: a case report. J Med Case Rep. 2017;11:263.PubMedPubMedCentralCrossRef Nakatani K, Kato T, Okada S, Matsumoto R, Nishida K, Komuro H, Suganuma T. Successful treatment with hyperbaric oxygen therapy for pneumatosis cystoides intestinalis as a complication of granulomatosis with polyangiitis: a case report. J Med Case Rep. 2017;11:263.PubMedPubMedCentralCrossRef
33.
go back to reference Deer M, Altorfer J, Pirovino M, Schmid M. Pneumatosis cystoides coli: a rare complication of colonoscopy. Endoscopy. 1983;15:119–20.CrossRef Deer M, Altorfer J, Pirovino M, Schmid M. Pneumatosis cystoides coli: a rare complication of colonoscopy. Endoscopy. 1983;15:119–20.CrossRef
34.
go back to reference Cyrany J, Kopácová M, Rejchrt S, Hornychová H, Tomsová M, Tycová V, Ryska A, Bures J. Puncture and cytology - sufficient for endoscopic diagnosis of pneumatosis cystoides intestinalis? Endoscopy. 2009;41(Suppl 2):E127–8.PubMedCrossRef Cyrany J, Kopácová M, Rejchrt S, Hornychová H, Tomsová M, Tycová V, Ryska A, Bures J. Puncture and cytology - sufficient for endoscopic diagnosis of pneumatosis cystoides intestinalis? Endoscopy. 2009;41(Suppl 2):E127–8.PubMedCrossRef
35.
go back to reference Takahashi K, Fujiya M, Ueno N, Ando K, Kashima S, Moriichi K, Okumura T. Endoscopic fine-needle aspiration is useful for the treatment of pneumatosis cystoides intestinalis with intussusception. Am J Gastroenterol. 2019;114(1):13.PubMedCrossRef Takahashi K, Fujiya M, Ueno N, Ando K, Kashima S, Moriichi K, Okumura T. Endoscopic fine-needle aspiration is useful for the treatment of pneumatosis cystoides intestinalis with intussusception. Am J Gastroenterol. 2019;114(1):13.PubMedCrossRef
36.
go back to reference Wang YJ, Wang YM, Zheng YM, Jiang HQ, Zhang J. Pneumatosis cystoides intestinalis: six case reports and a review of the literature. BMC Gastroenterol. 2018;18(1):100.PubMedPubMedCentralCrossRef Wang YJ, Wang YM, Zheng YM, Jiang HQ, Zhang J. Pneumatosis cystoides intestinalis: six case reports and a review of the literature. BMC Gastroenterol. 2018;18(1):100.PubMedPubMedCentralCrossRef
37.
go back to reference Kim YG, Kim KJ, Noh SH, Yang DH, Jung KW, Ye BD, Byeon JS, Myung SJ, Yang SK. Clear water filling and puncture: sufficient for endoscopic diagnosis of pneumatosis cystoides intestinalis? (with video). Gastrointest Endosc. 2011;74(5):1170–1.PubMedCrossRef Kim YG, Kim KJ, Noh SH, Yang DH, Jung KW, Ye BD, Byeon JS, Myung SJ, Yang SK. Clear water filling and puncture: sufficient for endoscopic diagnosis of pneumatosis cystoides intestinalis? (with video). Gastrointest Endosc. 2011;74(5):1170–1.PubMedCrossRef
38.
go back to reference Tahiri M, Levy J, Alzaid S, Anderson D. An approach to pneumatosis intestinalis: Factors affecting your management. Int J Surg Case Rep. 2015;6:133–137.CrossRef Tahiri M, Levy J, Alzaid S, Anderson D. An approach to pneumatosis intestinalis: Factors affecting your management. Int J Surg Case Rep. 2015;6:133–137.CrossRef
Metadata
Title
Pneumatosis cystoides intestinalis: a case report and literature review
Authors
Fangmei Ling
Di Guo
Liangru Zhu
Publication date
01-12-2019
Publisher
BioMed Central
Published in
BMC Gastroenterology / Issue 1/2019
Electronic ISSN: 1471-230X
DOI
https://doi.org/10.1186/s12876-019-1087-9

Other articles of this Issue 1/2019

BMC Gastroenterology 1/2019 Go to the issue
Live Webinar | 27-06-2024 | 18:00 (CEST)

Keynote webinar | Spotlight on medication adherence

Live: Thursday 27th June 2024, 18:00-19:30 (CEST)

WHO estimates that half of all patients worldwide are non-adherent to their prescribed medication. The consequences of poor adherence can be catastrophic, on both the individual and population level.

Join our expert panel to discover why you need to understand the drivers of non-adherence in your patients, and how you can optimize medication adherence in your clinics to drastically improve patient outcomes.

Prof. Kevin Dolgin
Prof. Florian Limbourg
Prof. Anoop Chauhan
Developed by: Springer Medicine
Obesity Clinical Trial Summary

At a glance: The STEP trials

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.

Developed by: Springer Medicine

Highlights from the ACC 2024 Congress

Year in Review: Pediatric cardiology

Watch Dr. Anne Marie Valente present the last year's highlights in pediatric and congenital heart disease in the official ACC.24 Year in Review session.

Year in Review: Pulmonary vascular disease

The last year's highlights in pulmonary vascular disease are presented by Dr. Jane Leopold in this official video from ACC.24.

Year in Review: Valvular heart disease

Watch Prof. William Zoghbi present the last year's highlights in valvular heart disease from the official ACC.24 Year in Review session.

Year in Review: Heart failure and cardiomyopathies

Watch this official video from ACC.24. Dr. Biykem Bozkurt discusses last year's major advances in heart failure and cardiomyopathies.