Abstract
Pneumatosis cystoid intestinalis (PCI) is a rare condition, with a worldwide incidence of 0.3–1.2%. PCI is classified into primary (idiopathic) and secondary forms, with 15% and 85% of presentations, respectively. This pathology was associated with a wide variety of underlining etiologies to explain the abnormal accumulation of gas within the submucosa (69.9%), subserosa (25.5%), or both layers (4.6%). Many patients endure misdiagnosis, mistreatment, or even inadequate surgical exploration. In this case, a patient presented acute diverticulitis, after treatment, a control colonoscopy was performed that found multiple rounds and elevated lesions. To further study the subepithelial lesion (SEL), a colorectal endoscopic ultrasound (EUS) was performed with an overtube in the same procedure. For safe insertion of the curvilinear array EUS, an overtube with colonoscopy was positioned through the sigmoid as described by Cheng et al. The EUS evaluation evidenced air reverberation in the submucosal layer. The pathological analysis was consistent with PCI’s diagnosis. The diagnosis of PCI is usually made by colonoscopy (51.9%), surgery (40.6%), and radiological findings (10.9%). Although the diagnosis can be made by radiological studies, a colorectal EUS and colonoscopy can be made in the same section without radiation and with high precision. As it is a rare disease, there are not enough studies to define the best approach, although colorectal EUS should be preferred for a reliable diagnosis.
Figure 1.
A 45-year-old female patient with a medical history of constipation and recurrent diverticulitis presented herself for an elective colonoscopy eight weeks after her last episode. In the descending colon the red arrows point to multiple rounded and elevated, softened smooth-surfaced, mucosal lesions with foci of enanthem were found without loss of mucosal contiguity. These lesions were homogeneously distributed over the entire circumference of this intestinal segment. PCI is classified into primary (idiopathic) and secondary forms, with 15% and 85% of presentations, respectively [1,2].
Figure 1.
A 45-year-old female patient with a medical history of constipation and recurrent diverticulitis presented herself for an elective colonoscopy eight weeks after her last episode. In the descending colon the red arrows point to multiple rounded and elevated, softened smooth-surfaced, mucosal lesions with foci of enanthem were found without loss of mucosal contiguity. These lesions were homogeneously distributed over the entire circumference of this intestinal segment. PCI is classified into primary (idiopathic) and secondary forms, with 15% and 85% of presentations, respectively [1,2].

Figure 2.
Although the diagnosis can be made by radiological studies [3,4,5,6], a colorectal EUS and colonoscopy can be made in the same section without radiation and with high precision [4,5,6]. An Endoscopic Ultrasound (EUS) (Video S1) was performed for lesion evaluation. The ultrasound images showed irregular hyperechoic structures, “dirty shadow”, A-lines, and decreased visualization of deeper structures, air reverberation compatible with gas in the submucosal layer, with no nodules or fluid inside.
Figure 2.
Although the diagnosis can be made by radiological studies [3,4,5,6], a colorectal EUS and colonoscopy can be made in the same section without radiation and with high precision [4,5,6]. An Endoscopic Ultrasound (EUS) (Video S1) was performed for lesion evaluation. The ultrasound images showed irregular hyperechoic structures, “dirty shadow”, A-lines, and decreased visualization of deeper structures, air reverberation compatible with gas in the submucosal layer, with no nodules or fluid inside.

Figure 3.
Biopsies of the subepithelial lesions were taken. Pathological analysis, found clusters of gas-filled cysts (as white balls) in the submucosa, and lamina propria were consistent with PCI’s diagnosis. The diverticulitis was the suspected reliable cause.
Figure 3.
Biopsies of the subepithelial lesions were taken. Pathological analysis, found clusters of gas-filled cysts (as white balls) in the submucosa, and lamina propria were consistent with PCI’s diagnosis. The diverticulitis was the suspected reliable cause.

Supplementary Materials
The following supporting information can be downloaded at: https://www.mdpi.com/article/10.3390/diagnostics13081424/s1, Video S1: Endoscopic Ultrasound View of Pneumatosis Cystoides Intestinalis.
Author Contributions
Conceptualization: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S. and E.G.H.d.M.; methodology: G.H.P.d.O. and J.G.R.J.S.; software: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S. and I.B.R.; validation: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S., M.B.B., M.K.M., I.B.R. and E.G.H.d.M.; formal analysis: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S., M.B.B., M.K.M., I.B.R. and E.G.H.d.M.; investigation: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S., M.B.B., M.K.M., I.B.R. and E.G.H.d.M.; resources: E.Y.Y., S.C., J.G.R.J.S., I.B.R. and E.G.H.d.M.; data curation: E.Y.Y., S.C., J.G.R.J.S. and I.B.R.; writing—original draft preparation: E.Y.Y., S.C. and J.G.R.J.S.; writing—review and editing: E.Y.Y., S.C., G.H.P.d.O., J.G.R.J.S., M.B.B., M.K.M., I.B.R. and E.G.H.d.M.; visualization, S.C.; supervision, S.C., I.B.R. and E.G.H.d.M.; project administration, S.C. and J.G.R.J.S.; funding acquisition: none. All authors have read and agreed to the published version of the manuscript.
Funding
This research received no external funding.
Institutional Review Board Statement
The study was conducted in accordance with the Declaration of Helsinki. Ethical review and approval were waived for this study by Ethics Committee of Universidade Sao Paulo. due to the reason of “Retrospectively performed scan image without research intervention. The patient signed an informed consent form prior to the examination”.
Informed Consent Statement
Written informed consent for the case to be published (incl. images, case history, and data) was obtained from the patient.
Data Availability Statement
No new data were created or analyzed in this study. Data sharing is not applicable to this article.
Conflicts of Interest
Eduardo Guimarães Hourneaux de Moura reports personal fees from Boston Scientific and Olympus, but these were not relevant to this manuscript. The other authors declare no conflict of interest.
References
- Piltcher-da-Silva, R.; Sasaki, V.L.; Zangari, M.A.C.; Gallotti, F.M.; Saenger, B.F.; Piltcher-Recuero, M.; de Melo Rocha, G.; da Costa, M.R.; Coelho, J.C.U. Pneumatosis cystoides intestinalis with pneumoperitoneum in an 87-years-old male patient: A case report. J. Surg. Case Rep. 2022, 2022, rjac103. [Google Scholar] [CrossRef] [PubMed]
- Wu, L.L.; Yang, Y.S.; Dou, Y.; Liu, Q.S. A systematic analysis of pneumatosis cystoids intestinalis. World J. Gastroenterol. 2013, 19, 4973–4978. [Google Scholar] [CrossRef] [PubMed]
- Cheng, S.P.; Matuguma, S.E.M.; de Oliveira, G.H.P.M.; Silva, G.L.M.; Cheng, H.P.; Sánchez-Luna, S.A.M.; Minata, M.K.M. Colonoscopic Ultrasound-Guided Fine-Needle Aspiration Using a Curvilinear Array Transducer: A Single-Center Retrospective Cohort Study. Dis. Colon. Rectum. 2022, 65, e80–e84. [Google Scholar] [CrossRef] [PubMed]
- Castro-Poças, F.; Araújo, T.; Pedroto, I. Endoscopic ultrasound of pneumatosis cystoides intestinalis. Endoscopy 2015, 47 (Suppl 1), E274. [Google Scholar] [CrossRef] [PubMed]
- Ling, F.; Guo, D.; Zhu, L. Pneumatosis cystoides intestinalis: A case report and literature review. BMC Gastroenterol. 2019, 19, 176. [Google Scholar] [CrossRef] [PubMed]
- Shimada, M.; Ina, K.; Takahashi, H.; Horiuchi, Y.; Imada, A.; Nishio, Y.; Ando, T.; Kusugami, K. Pneumatosis cystoides intestinalis treated with hyperbaric oxygen therapy: Usefulness of an endoscopic ultrasonic catheter probe for diagnosis. Intern Med. 2001, 40, 896–900. [Google Scholar] [CrossRef] [PubMed]
Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury to people or property resulting from any ideas, methods, instructions or products referred to in the content. |
© 2023 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).