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Abstract

Peritoneal inclusion cysts (PICs) are a rare and benign condition of uncertain pathogenesis. The fluid-filled, mesothelial-lined cysts manifest within the abdominopelvic cavity. This case report details an unusual occurrence of a 97 mm PIC- presenting as an umbilical hernia- in a 26-year-old male patient with no prior surgical history. Following pre-operative cross-sectional imaging, this was managed through open excision without complication. A systematic review of the literature highlighted 30 previous cases [26F, 4M] with a mean age of 34 years (std ±15.4) and a median diameter of 93 mm [IQR, 109 mm]. A total of 53% (n = 16) of cases had a history of previous abdominal surgery. Surgical excision is safe and laparoscopic modality should be considered (<1% recurrence). Accepting the limited evidence base, image guided drainage should be avoided (50% recurrence, n = 2).

peritoneal inclusion cyst, multicystic peritoneal mesothelioma, umbilical hernia

Introduction

Peritoneal inclusion cysts (PICs), also known as multicystic peritoneal mesotheliomas, are a rare and benign condition of uncertain pathogenesis [1]. The fluid-filled, mesothelial lined cysts manifest within the abdominopelvic cavity and can vary in size. In the few case reports presented, they are predominantly observed in women of childbearing age and have been associated with chronic peritoneal inflammation stemming from a variety of aetiologies and prior surgical interventions [2, 3]. To date, surgical excision has been the only considered definitive treatment [4]. This paper details a case of PIC presenting as an umbilical hernia, reported as per the for CAse REports (CARE) guidelines. [5], and a systematic review of the literature compliant with Preferred Reporting Items for Systematic Review Meta-Analysis (PRISMA) guidelines [6].

Case report

We present the case of a 26-year-old man who presented to the general surgery outpatient clinic with intermittent peri-umbilical pain, and the presence of a soft swelling palpable at his umbilicus. There was a positive cough impulse consistent with a true umbilical hernia and the patient described no previous medical or surgical history. An abdominal ultrasound scan visualized a large thin-walled serous fluid collection tracking into the peritoneal cavity (95 mm × 87 mm × 97 mm) (Fig. 1). Computed tomography (CT) of the abdomen and pelvis with contrast reported a large bi-lobed cystic mass centred at the right side of the mesentery, with herniation of part of the cyst along the umbilicus. Displacement of the small bowel with anterior extension into the abdominal wall was seen with the suggestion of posterior extension into the right retroperitoneal space (Figs 2 and 3). The patient underwent routine pre-operative work up and the cyst was excised through a midline para-umbilical laparotomy (Fig. 4) given the concern of retroperitoneal involvement. Intra-operatively, the hernia neck and the root of the cyst were found at the base of the umbilical cicatrix with no attachment to the mesentery. The cyst was loculated and filled with clear fluid. It was dissected off the peritoneal tissues and off from the posterior umbilical skin prior to removal. There was evidence of rupture of one of the locules and clear fluid was drained.

Ultrasound image demonstrating fluid collection within the umbilicus (a) tracking into abdominal cavity (c) originating from the cicatrix (b).
Figure 1

Ultrasound image demonstrating fluid collection within the umbilicus (a) tracking into abdominal cavity (c) originating from the cicatrix (b).

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CT scan demonstrating bilobed cystic mass.
Figure 2

CT scan demonstrating bilobed cystic mass.

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CT scan in sagittal cross-section demonstrating PIC.
Figure 3

CT scan in sagittal cross-section demonstrating PIC.

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PIC was excised through midline laparotomy given the suggestion of retroperitoneal involvement on pre-operative imaging.
Figure 4

PIC was excised through midline laparotomy given the suggestion of retroperitoneal involvement on pre-operative imaging.

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Histological examination demonstrated a paucicellular fibrous wall including frequent small blood vessels. The outer surface constitutionally comprised of loose fibrous tissue mixed with adipose tissue, and the lining was composed of a monolayer and- focally- a double layer of mesothelial cells. This showed no significant atypia, and a diagnosis of PIC with no evidence of neoplasia was made. He was discharged without complication, with a follow up CT planned at 6 months.

Systematic search

The MEDLINE, EMBASE electronic databases published between 1 January 1947 and 1January 2024 were searched using the term ‘peritoneal inclusion cyst’. All studies which reported the case of a PIC were considered. KN independently retrieved article abstracts with JB cross-checking. The full texts of potentially eligible studies were retrieved and independently assessed for eligibility by KN and JB according to predefined inclusion criteria. Any disagreement was resolved through discussion with PG. Initial search yielded 61 full text articles from 1946 to 2023 for review as displayed in the PRISMA Flow Chart (Fig. 5), with 30 included in the final analysis (Table 1).

Preferred Reporting Items for Systematic Review Meta-Analysis (PRISMA) flow chart showing selection of reports for discussion.
Figure 5

Preferred Reporting Items for Systematic Review Meta-Analysis (PRISMA) flow chart showing selection of reports for discussion.

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Table 1
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Systematic search.

Author(s)CountrySexAgeMax.Diameter (mm)ImagingOriginInterventionComplicationsPrevious surgery
Coulibaly et al. 2023 [7]FranceFemale29-PETPelvic cavityLaparoscopicNoneNot documented
AlTamimi et al. 2023 [3]United StatesFemale18150CTOvaryOpenFailed IR drainagePrevious open bladder repair
Guo et al. 2023 [8]EnglandFemale4373USSVaginal/uterine spaceOpenNonePrevious fibroid and ovarian cyst removal. Previous appendicectomy
Ivanova et al. 2022 [9]Russian FederationFemale---OvaryLaparoscopic-Not documented
Subramonian et al. 2021 [10]NetherlandsFemale1487USSRecto-vaginal spaceLaparoscopicNonePrevious laparotomy (ileal perforation)
Cosgrove et al. 2021 [11]Northern IrelandMale4186CT/MRIMesentery to the sigmoid colonOpenNoneNot documented
Katebi et al. 2021 [12]NetherlandsFemale44200CTRecto-vaginal spaceLaparoscopicNoneNot documented
Kato et al. 2010 [13]JapanFemale21-Not recordedPelvic cavityNot documentedNot recordedNot documented
Wolf et al. 2020 [14]NetherlandsFemale72-CT/MRIPelvic cavityOpenRecurrencePrevious hysterectomy
Napoe and Rardin 2020 [15]EnglandFemale4750CTUterineOpenNoneHysterectomy
Katebi et al. 2019 [12]NetherlandsFemale44200MRIRecto-vaginal spaceLaparoscopicNot recordedNot documented
Pereira 2019 [16]EnglandFemale34107MRIPelvic cavitySpontaneously rupturedNonePrevious robotic assisted endometrial resection, appendicectomy and adhesiolysis
Mishra et al. 2018 [17]IndiaMale43210USS/CTMesocolonLaparoscopicNoneNone
De Luca et al. 2017 [18]NetherlandsMale4980USSMesenteric root / precavalLaparoscopicNonePrevious open appendicectomy
Sato et al. 2017 [19]IrelandFemale---OvaryLaparoscopic-2 prior laparotomies
Singh et al. 2015 [2]IndiaFemale--CTOvaryOpenNonePrevious bilateral tubal ligation
Trehan. 2014 [20]EnglandFemale5157MRIRecto-vaginal spaceLaparoscopicNonePrevious bilateral tubal ligation
Kanasugi et al. 2013 [21]JapanFemale31-MRI/USSPelvic cavityOpenNoneNot documented
Saxena et al. 2011 [22]United StatesFemale780USS/MRIAscending colon mesenteryLaparoscopicNoneNot documented
Ho-Fung et al. 2011 [23]United StatesFemale--Pelvic cavityCystocentesisNoneNot documented
Dillman and DiPietro 2009 [24]GermanyFemale16-USSOvaryLaparoscopicNoneMultiple previous adhesiolysis
Vallerie et al. 2008 [25]United StatesFemale29217USSPelvic cavityCystocentesisRecurrence2 previous exploratory laparotomies
Advincula and Hernandez 2006 [26]United StatesFemale36100CTRecto-vaginal spaceLaparoscopicNonePrevious hysterectomy and exploratory laparotomy
Phupong et al. 2005 [27]GermanyFemale3361USSOvaryLaparoscopicNoneNot documented
Nayak et al. 2005 [28]IndiaFemale260Found during caesareanPelvic cavityOpenNonePrevious caesarean section
Durak et al. 2005 [29]United StatesMale32140CTPelvic cavityOpenNot documented
Toprak et al. 2004 [30]United StatesFemale24-USS/CT/MRIAdnexaOpenNoneNone
Omeroglu and Husain. 2001 [31]United StatesFemale3175CTOvarianOpenNonePrevious myomectomy
Brustmann 2000 [32]United StatesFemale21200-Mesentery of terminal ileumOpenNoneNot documented
Lamovec and Sinkovec 1996 [33]EnglandFemale68200CT/MRIMesentry of descending colonOpenNoneNot documented
Author(s)CountrySexAgeMax.Diameter (mm)ImagingOriginInterventionComplicationsPrevious surgery
Coulibaly et al. 2023 [7]FranceFemale29-PETPelvic cavityLaparoscopicNoneNot documented
AlTamimi et al. 2023 [3]United StatesFemale18150CTOvaryOpenFailed IR drainagePrevious open bladder repair
Guo et al. 2023 [8]EnglandFemale4373USSVaginal/uterine spaceOpenNonePrevious fibroid and ovarian cyst removal. Previous appendicectomy
Ivanova et al. 2022 [9]Russian FederationFemale---OvaryLaparoscopic-Not documented
Subramonian et al. 2021 [10]NetherlandsFemale1487USSRecto-vaginal spaceLaparoscopicNonePrevious laparotomy (ileal perforation)
Cosgrove et al. 2021 [11]Northern IrelandMale4186CT/MRIMesentery to the sigmoid colonOpenNoneNot documented
Katebi et al. 2021 [12]NetherlandsFemale44200CTRecto-vaginal spaceLaparoscopicNoneNot documented
Kato et al. 2010 [13]JapanFemale21-Not recordedPelvic cavityNot documentedNot recordedNot documented
Wolf et al. 2020 [14]NetherlandsFemale72-CT/MRIPelvic cavityOpenRecurrencePrevious hysterectomy
Napoe and Rardin 2020 [15]EnglandFemale4750CTUterineOpenNoneHysterectomy
Katebi et al. 2019 [12]NetherlandsFemale44200MRIRecto-vaginal spaceLaparoscopicNot recordedNot documented
Pereira 2019 [16]EnglandFemale34107MRIPelvic cavitySpontaneously rupturedNonePrevious robotic assisted endometrial resection, appendicectomy and adhesiolysis
Mishra et al. 2018 [17]IndiaMale43210USS/CTMesocolonLaparoscopicNoneNone
De Luca et al. 2017 [18]NetherlandsMale4980USSMesenteric root / precavalLaparoscopicNonePrevious open appendicectomy
Sato et al. 2017 [19]IrelandFemale---OvaryLaparoscopic-2 prior laparotomies
Singh et al. 2015 [2]IndiaFemale--CTOvaryOpenNonePrevious bilateral tubal ligation
Trehan. 2014 [20]EnglandFemale5157MRIRecto-vaginal spaceLaparoscopicNonePrevious bilateral tubal ligation
Kanasugi et al. 2013 [21]JapanFemale31-MRI/USSPelvic cavityOpenNoneNot documented
Saxena et al. 2011 [22]United StatesFemale780USS/MRIAscending colon mesenteryLaparoscopicNoneNot documented
Ho-Fung et al. 2011 [23]United StatesFemale--Pelvic cavityCystocentesisNoneNot documented
Dillman and DiPietro 2009 [24]GermanyFemale16-USSOvaryLaparoscopicNoneMultiple previous adhesiolysis
Vallerie et al. 2008 [25]United StatesFemale29217USSPelvic cavityCystocentesisRecurrence2 previous exploratory laparotomies
Advincula and Hernandez 2006 [26]United StatesFemale36100CTRecto-vaginal spaceLaparoscopicNonePrevious hysterectomy and exploratory laparotomy
Phupong et al. 2005 [27]GermanyFemale3361USSOvaryLaparoscopicNoneNot documented
Nayak et al. 2005 [28]IndiaFemale260Found during caesareanPelvic cavityOpenNonePrevious caesarean section
Durak et al. 2005 [29]United StatesMale32140CTPelvic cavityOpenNot documented
Toprak et al. 2004 [30]United StatesFemale24-USS/CT/MRIAdnexaOpenNoneNone
Omeroglu and Husain. 2001 [31]United StatesFemale3175CTOvarianOpenNonePrevious myomectomy
Brustmann 2000 [32]United StatesFemale21200-Mesentery of terminal ileumOpenNoneNot documented
Lamovec and Sinkovec 1996 [33]EnglandFemale68200CT/MRIMesentry of descending colonOpenNoneNot documented
Table 1
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Systematic search.

Author(s)CountrySexAgeMax.Diameter (mm)ImagingOriginInterventionComplicationsPrevious surgery
Coulibaly et al. 2023 [7]FranceFemale29-PETPelvic cavityLaparoscopicNoneNot documented
AlTamimi et al. 2023 [3]United StatesFemale18150CTOvaryOpenFailed IR drainagePrevious open bladder repair
Guo et al. 2023 [8]EnglandFemale4373USSVaginal/uterine spaceOpenNonePrevious fibroid and ovarian cyst removal. Previous appendicectomy
Ivanova et al. 2022 [9]Russian FederationFemale---OvaryLaparoscopic-Not documented
Subramonian et al. 2021 [10]NetherlandsFemale1487USSRecto-vaginal spaceLaparoscopicNonePrevious laparotomy (ileal perforation)
Cosgrove et al. 2021 [11]Northern IrelandMale4186CT/MRIMesentery to the sigmoid colonOpenNoneNot documented
Katebi et al. 2021 [12]NetherlandsFemale44200CTRecto-vaginal spaceLaparoscopicNoneNot documented
Kato et al. 2010 [13]JapanFemale21-Not recordedPelvic cavityNot documentedNot recordedNot documented
Wolf et al. 2020 [14]NetherlandsFemale72-CT/MRIPelvic cavityOpenRecurrencePrevious hysterectomy
Napoe and Rardin 2020 [15]EnglandFemale4750CTUterineOpenNoneHysterectomy
Katebi et al. 2019 [12]NetherlandsFemale44200MRIRecto-vaginal spaceLaparoscopicNot recordedNot documented
Pereira 2019 [16]EnglandFemale34107MRIPelvic cavitySpontaneously rupturedNonePrevious robotic assisted endometrial resection, appendicectomy and adhesiolysis
Mishra et al. 2018 [17]IndiaMale43210USS/CTMesocolonLaparoscopicNoneNone
De Luca et al. 2017 [18]NetherlandsMale4980USSMesenteric root / precavalLaparoscopicNonePrevious open appendicectomy
Sato et al. 2017 [19]IrelandFemale---OvaryLaparoscopic-2 prior laparotomies
Singh et al. 2015 [2]IndiaFemale--CTOvaryOpenNonePrevious bilateral tubal ligation
Trehan. 2014 [20]EnglandFemale5157MRIRecto-vaginal spaceLaparoscopicNonePrevious bilateral tubal ligation
Kanasugi et al. 2013 [21]JapanFemale31-MRI/USSPelvic cavityOpenNoneNot documented
Saxena et al. 2011 [22]United StatesFemale780USS/MRIAscending colon mesenteryLaparoscopicNoneNot documented
Ho-Fung et al. 2011 [23]United StatesFemale--Pelvic cavityCystocentesisNoneNot documented
Dillman and DiPietro 2009 [24]GermanyFemale16-USSOvaryLaparoscopicNoneMultiple previous adhesiolysis
Vallerie et al. 2008 [25]United StatesFemale29217USSPelvic cavityCystocentesisRecurrence2 previous exploratory laparotomies
Advincula and Hernandez 2006 [26]United StatesFemale36100CTRecto-vaginal spaceLaparoscopicNonePrevious hysterectomy and exploratory laparotomy
Phupong et al. 2005 [27]GermanyFemale3361USSOvaryLaparoscopicNoneNot documented
Nayak et al. 2005 [28]IndiaFemale260Found during caesareanPelvic cavityOpenNonePrevious caesarean section
Durak et al. 2005 [29]United StatesMale32140CTPelvic cavityOpenNot documented
Toprak et al. 2004 [30]United StatesFemale24-USS/CT/MRIAdnexaOpenNoneNone
Omeroglu and Husain. 2001 [31]United StatesFemale3175CTOvarianOpenNonePrevious myomectomy
Brustmann 2000 [32]United StatesFemale21200-Mesentery of terminal ileumOpenNoneNot documented
Lamovec and Sinkovec 1996 [33]EnglandFemale68200CT/MRIMesentry of descending colonOpenNoneNot documented
Author(s)CountrySexAgeMax.Diameter (mm)ImagingOriginInterventionComplicationsPrevious surgery
Coulibaly et al. 2023 [7]FranceFemale29-PETPelvic cavityLaparoscopicNoneNot documented
AlTamimi et al. 2023 [3]United StatesFemale18150CTOvaryOpenFailed IR drainagePrevious open bladder repair
Guo et al. 2023 [8]EnglandFemale4373USSVaginal/uterine spaceOpenNonePrevious fibroid and ovarian cyst removal. Previous appendicectomy
Ivanova et al. 2022 [9]Russian FederationFemale---OvaryLaparoscopic-Not documented
Subramonian et al. 2021 [10]NetherlandsFemale1487USSRecto-vaginal spaceLaparoscopicNonePrevious laparotomy (ileal perforation)
Cosgrove et al. 2021 [11]Northern IrelandMale4186CT/MRIMesentery to the sigmoid colonOpenNoneNot documented
Katebi et al. 2021 [12]NetherlandsFemale44200CTRecto-vaginal spaceLaparoscopicNoneNot documented
Kato et al. 2010 [13]JapanFemale21-Not recordedPelvic cavityNot documentedNot recordedNot documented
Wolf et al. 2020 [14]NetherlandsFemale72-CT/MRIPelvic cavityOpenRecurrencePrevious hysterectomy
Napoe and Rardin 2020 [15]EnglandFemale4750CTUterineOpenNoneHysterectomy
Katebi et al. 2019 [12]NetherlandsFemale44200MRIRecto-vaginal spaceLaparoscopicNot recordedNot documented
Pereira 2019 [16]EnglandFemale34107MRIPelvic cavitySpontaneously rupturedNonePrevious robotic assisted endometrial resection, appendicectomy and adhesiolysis
Mishra et al. 2018 [17]IndiaMale43210USS/CTMesocolonLaparoscopicNoneNone
De Luca et al. 2017 [18]NetherlandsMale4980USSMesenteric root / precavalLaparoscopicNonePrevious open appendicectomy
Sato et al. 2017 [19]IrelandFemale---OvaryLaparoscopic-2 prior laparotomies
Singh et al. 2015 [2]IndiaFemale--CTOvaryOpenNonePrevious bilateral tubal ligation
Trehan. 2014 [20]EnglandFemale5157MRIRecto-vaginal spaceLaparoscopicNonePrevious bilateral tubal ligation
Kanasugi et al. 2013 [21]JapanFemale31-MRI/USSPelvic cavityOpenNoneNot documented
Saxena et al. 2011 [22]United StatesFemale780USS/MRIAscending colon mesenteryLaparoscopicNoneNot documented
Ho-Fung et al. 2011 [23]United StatesFemale--Pelvic cavityCystocentesisNoneNot documented
Dillman and DiPietro 2009 [24]GermanyFemale16-USSOvaryLaparoscopicNoneMultiple previous adhesiolysis
Vallerie et al. 2008 [25]United StatesFemale29217USSPelvic cavityCystocentesisRecurrence2 previous exploratory laparotomies
Advincula and Hernandez 2006 [26]United StatesFemale36100CTRecto-vaginal spaceLaparoscopicNonePrevious hysterectomy and exploratory laparotomy
Phupong et al. 2005 [27]GermanyFemale3361USSOvaryLaparoscopicNoneNot documented
Nayak et al. 2005 [28]IndiaFemale260Found during caesareanPelvic cavityOpenNonePrevious caesarean section
Durak et al. 2005 [29]United StatesMale32140CTPelvic cavityOpenNot documented
Toprak et al. 2004 [30]United StatesFemale24-USS/CT/MRIAdnexaOpenNoneNone
Omeroglu and Husain. 2001 [31]United StatesFemale3175CTOvarianOpenNonePrevious myomectomy
Brustmann 2000 [32]United StatesFemale21200-Mesentery of terminal ileumOpenNoneNot documented
Lamovec and Sinkovec 1996 [33]EnglandFemale68200CT/MRIMesentry of descending colonOpenNoneNot documented

Discussion

This is the first documented case of a PIC originating from the umbilical cicatrix. Thirty reported cases of PIC have been identified, 26 female and 4 male, with a mean age at presentation of 34 years (std ±15.4) and a median diameter of 93 mm [IQR, 109 mm]. A total of 53% (n = 16) of cases had a history of a previous abdominal surgery. Thirteen were excised via laparotomy, 13 were excised laparoscopically, 2 were drained under Interventional radiology guidance, 1 ruptured spontaneously with no further sequalae, and 1 case did not report their operative approach. Of the two cases that were drained, one recurred. Of those excised, only one recurred (median follow up 24 months (range 0–60 months)).

The most common organ of origin described was ovarian- in 23% of cases (N = 7), followed by the recto-vaginal space (17%, n = 5). In seven cases (23%) the origin was broadly defined as the pelvic cavity. Other origins included mesentery of small and large bowel, adnexa, and the vaginal-uterine space.

This case underscores the atypical presentation of a PIC in a surgically naïve male patient, emphasisizing the importance of pre-operative imaging in those presenting with atypical features of hernia; this instance, skin changes and discharge in the absence of previous abdominal surgery. Existing literature supports surgical excision rather than drainage given the risk of recurrence. Surveillance and required follow up specificities are currently unknown and further research is needed to determine this.

Author contributions

KN completed the data collection, statistical analysis of literature and drafting of manuscript. EC, PG & JB contributed to case conception, and manuscript editing.

Conflict of interest statement

The authors declare no conflicts of interest.

Funding

None declared.

Consent

Informed patient consent was obtained and no identifiable details are mentioned in this report.

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Published by Oxford University Press and JSCR Publishing Ltd. © The Author(s) 2024.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited.
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Subject
Colorectal Surgery
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Case Report
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