Recurrent obturator hernia in a patient with autosomal dominant polycystic kidney disease undergoing peritoneal dialysis: a case report

Abstract

Obturator hernia, a rare condition that accounts for only 0.07–1% of all hernias, typically occurs in older adult, thin, multiparous women owing to weakening of the obturator membrane and increased intra-abdominal pressure. Conditions such as chronic constipation, chronic obstructive pulmonary disease, ascites, peritoneal dialysis (PD), and autosomal dominant polycystic kidney disease (ADPKD) are considered risk factors for an obturator hernia. Diagnosis is often delayed because of nonspecific symptoms, and computed tomography imaging is essential for early detection. We report the case of a 69-year-old woman with ADPKD undergoing PD who developed a right-sided obturator hernia, followed by contralateral recurrences within 3 months. The initial and recurrent hernias were managed with open surgical repair using simple suture techniques. The patient's enlarged kidneys and liver, combined with dialysate fluid, may have contributed to sustained elevated intra-abdominal pressure, increasing the risk for hernia recurrence. This case highlights the unique combination of ADPKD and PD as potential contributors to obturator hernia recurrence.

Introduction

Obturator hernia is a rare disease that accounts for only 0.07–1% of all hernias and is frequently seen in older adult, thin, multiparous women [1]. It occurs when the canal is enlarged, and a hernia sac is formed because of weakening of the obturator membrane [2]. Chronic obstructive pulmonary disease, chronic constipation, kyphoscoliosis, pregnancy, and ascites are risk factors for obturator hernia because of the increased intra-abdominal pressure (IAP) in these conditions [1]. Acute mechanical bowel obstruction and abdominal pain are the common symptoms of an obturator hernia. However, there are no specific clinical features or physical examination findings, and a computed tomography (CT) scan is usually necessary for diagnosis [1, 2]. An obturator hernia has a high morbidity and mortality rate (50–70%); therefore, early surgical treatment is required after detection [1]. The recurrence rate is 2–10%, depending on the operative approaches and surgical techniques [3, 4].

We present the case of a patient with an obturator hernia and Autosomal dominant polycystic kidney disease (ADPKD) who was on peritoneal dialysis (PD) and experienced recurrences following surgery.

Case report

A 69-year-old woman presented with nausea and right groin pain after having spent the prior day in a standing position for an extended time. She was diagnosed with ADPKD at 38 years of age, with a complication of polycystic liver (Fig. 1a). As her kidney function worsened, renal replacement therapy became necessary. Although her ejection fraction on echocardiography was preserved, she had undergone surgery for aortic and mitral valve regurgitation 5 years earlier, raising concern for the risk of heart failure following initiation of hemodialysis (HD). Moreover, she wished to maintain greater daytime freedom. After discussing the risks and benefits of initiating PD in accordance with clinical guidelines, PD was initiated 1 year earlier. The patient was undergoing continuous cycling PD with four exchanges per day—three with 1500 mL of 1.5% glucose at night and one with 1,000 mL of icodextrin during the day. She also had a history of a uterine prolapse and three prior deliveries.

Fig. 1.

a Contrast-enhanced CT of the abdomen showing autosomal dominant polycystic kidney disease and polycystic liver. b Contrast-enhanced CT of the abdomen showing a right obturator hernia (arrowhead). c Contrast-enhanced CT of the abdomen showing a left obturator hernia (arrowhead). d Contrast-enhanced CT of the abdomen showing a left obturator hernia (arrowhead)

Physical examination revealed a height of 149.5 cm, body weight of 45.5 kg, and body mass index of 20.4 kg/m². Her vital signs showed a temperature of 36.5℃, blood pressure of 156/95 mmHg, pulse rate of 89 beats per min, and respiration rate of 18 breaths per min. The conjunctiva bulbi was not icteric. Heart sounds were normal, and no murmur was detected. Auscultation revealed clear bilateral lung sounds and no rales. The abdomen was soft, and bowel sounds were normal. Right groin tenderness was present; however, muscular rigidity was absent. There was no redness, tenderness, or drainage of pus at the PD catheter exit site. The rest of the physical examination was unremarkable. Laboratory tests showed a white blood cell count of 9300/μL with 91% neutrophils, and C-reactive protein level of 0.07 mg/dL. A chest radiograph showed no infiltration shadows or pleural effusion. A contrast-enhanced CT image of the abdomen revealed a right incarcerated obturator hernia, with no hernia detected on the left side (Fig. 1b). The total kidney volume was 4329 mL (right: 2,178 mL; left: 2,200 mL), and the liver cyst volume was 767 mL (measured using the ellipsoid method). Emergency open surgical repair (simple suture repair) was performed on the same day. After surgery, PD was discontinued, and HD was initiated through a central venous catheter in her left forearm, considering her psychological burden. As time was needed before starting to use the left forearm shunt, ambulatory PD was performed with three exchanges of 1,000 mL of 1.5% glucose per day, 19 days after the right obturator hernia repair surgery. The patient was discharged until HD was started. She did not report any abdominal symptoms after restarting PD.

She presented again with nausea and right groin pain, just 2 days after discharge and 8 days after restarting PD. Laboratory tests showed a white blood cell count of 5700/μL with 81.6% neutrophils and C-reactive protein level of 0.12 mg/dL. Contrast-enhanced CT imaging of the abdomen revealed an incarcerated left obturator hernia, with no hernia detected on the right side (Fig. 1c). The total kidney volume was 4,396 mL (right: 2232 mL; left: 2,164 mL), and the liver cyst volume was 800 mL (measured using the ellipsoid method). Emergency open surgical repair (simple suture repair) was performed. Continuation of PD was considered too difficult for the patient, so the peritoneal catheter was removed. She was discharged after the initiation of HD using her left forearm shunt.

However, the patient again presented with the same symptoms 3 months after the second discharge and was diagnosed with a left obturator hernia using contrast-enhanced CT (Fig. 1d). The total kidney volume was 3644 mL (right: 1666 mL; left: 1978 mL), and the liver cyst volume was 785 mL (measured using the ellipsoid method). Laboratory tests showed a white blood cell count of 6980/μL with 88.7% neutrophils and C-reactive protein level of 0.06 mg/dL. This time, emergency open surgical repair (simple suture repair) was performed, and her uterus was repositioned by bending it forward to prevent the intestinal tract from falling into the vesicouterine pouch or rectouterine pouch. The patient’s condition improved, with no recurrence of the obturator hernia at the 6-month follow-up.

Discussion

PD increases IAP through the instillation of dialysates into the peritoneal cavity, making abdominal hernia a potential complication of the procedure [5, 6]. The prevalence of abdominal hernias in patients undergoing PD is 7–25% [7], with incisional hernias, hernias at the catheter placement site, inguinal hernias, and umbilical hernias being the most common types [8].

In contrast, obturator hernias are exceedingly rare in patients undergoing PD, with only three cases reported to date [9–11]. Furthermore, there have been no reported cases of recurrence.

In our case, the patient experienced the first recurrence of an obturator hernia 1 month after her initial surgical repair, and the second recurrence occurred 3 months later (Table 1). Although information regarding the cause of obturator hernia recurrence is limited, we present two possible reasons.

Table 1.

Cases of obturator hernia with chronic kidney disease on peritoneal dialysis [10–12]

	Case 1 [10]	Case 2 [11]	Case 3 [12]	Our case
Age (years)	58	82	72	69
Sex	Woman	Woman	Woman	Woman
Cause of CKD	Hepatitis B and C viral infection	Hypertensive nephrosclerosis	Diabetic nephropathy	ADPKD
Type of PD	CAPD	CAPD	N/A
Site of Hernia	Right	Bilateral	N/A	Right
Symptom	1-year history of right proximal medial thigh mass	5-month history of severe bilateral thigh pain and swelling	None	Right groin tenderness
Treatment	Open surgery Mesh repair	Laparoscopy Mesh repair	Open surgery partial resection of the ileum, divided ileostomy, and the creation of a mucus fistula	Open surgery Simple suture
Recurrence	None	None	N/A	Left

CKD chronic kidney disease, ADPKD autosomal dominant polycystic kidney disease, PD peritoneal dialysis; CAPD, continuous ambulatory peritoneal dialysis

10. Kao SY, Lee TC, Weng ZC, Chen TH, Tsai PJ. Treatment of Obturator Hernia in a Patient Undergoing Peritoneal Dialysis. Perit Dial Int. 2014; 34:803. https://doi.org/10.3747/PDI.2013.00116

11. Ramkumar J, Lu D, Scott T. Laparoscopic Mesh Repair of Bilateral Obturator Hernias Post-Peritoneal Dialysis. Perit Dial Int. 2019; 39:95–7. https://doi.org/10.3747/PDI.2018.00203

12. Arata R, Banshodani M, Yamashita M, Shintaku S, Moriishi M, Kawanishi H. Perforative peritonitis confused with peritoneal dialysis-related peritonitis: Report of three cases. Int J Surg Case Rep. 2020; 70:20–3. https://doi.org/10.1016/J.IJSCR.2020.03.046

First, this patient was undergoing PD and had a history of ADPKD. Patients with ADPKD who are on PD have a higher risk of abdominal hernia compared to patients without ADPKD undergoing PD [12]. ADPKD itself predisposes individuals to abdominal hernia because of abnormalities in the production of the basement membrane or extracellular matrix [13]. Furthermore, some studies have reported that the presence of ADPKD, in addition to PD, contributes to further increases in IAP, leading to the development of abdominal hernias [12].

Although a previous study found no association between organ volume and IAP in patients undergoing PD [14], in our patient, the total kidney volume was 4329 mL, the liver cyst volume was 767 mL, and the peritoneal dialysate volume was 1000–1500 mL. The organ volumes reported in that study were smaller than those observed in the present case, and in studies including patients with organ volumes comparable to or larger than those observed here, PD leakage and abdominal hernias have been reported [15]. These findings suggest that large organ volume may have contributed to increased IAP in this case.

Furthermore, previous reports have shown that kidney and liver volumes may increase after initiation of PD in patients with ADPKD [16]. In our patient, at the time of recurrence after continued PD, the total kidney volume had increased to 4,396 mL, whereas the liver cyst volume remained 800 mL. This increase in organ volume may have further elevated IAP and contributed to hernia recurrence.

A second possible reason for the recurrence of an obturator hernia was that the patient underwent open surgery, and a simple suture closure was chosen to repair the obturator hernia.

In general, the surgical approaches for obturator hernia include open surgery or laparoscopic surgery, with either mesh repair or suture repair [3]. Compared with open surgery, laparoscopic treatment has advantages because of its minimal invasiveness, wide visual field, and a lower recurrence rate [3, 4]. Mesh repair is also associated with a lower recurrence rate regardless of the surgical approach [4].

Although there are no studies directly comparing laparoscopic and open hernia repair specifically in patients with ADPKD, mesh-based repair has been shown to reduce recurrence risk and is recommended even in this population [17]. In patients undergoing PD, mesh repair has also been associated with lower recurrence rates [18], and laparoscopic hernia repair during PD has been reported [19]. Therefore, laparoscopic mesh repair may be considered a treatment option for obturator hernia in patients with ADPKD undergoing PD.

In our case, there was a history of prior abdominal surgery for the insertion of a PD catheter, in addition to the patient having ADPKD, which might have led to adhesions in the intra-abdominal tissues, making it difficult to secure the surgical field in a laparoscopic approach. Moreover, given the potential for hernia incarceration and bowel necrosis, we opted for open surgery and decided not to use a mesh in the procedure because of the risk of infection. However, the surgical approach and repair method may have contributed to recurrence.

Several strategies may be considered to prevent the development and recurrence of hernias in patients with ADPKD. First, careful consideration is required when selecting PD as a dialysis modality in patients with ADPKD. The 2025 KDIGO ADPKD guideline states that there is no difference in mortality between HD and PD in patients with ADPKD and that the choice of dialysis modality should be based on patient-related factors, patient preference, and availability of facilities. However, in addition to general contraindications for PD, the guideline emphasizes that marked enlargement of the kidneys and/or liver warrants careful consideration before initiating PD [20]. In the present case, the indication for PD was determined according to the then-current 2014 guideline in Japan, which did not explicitly state that marked renal and/or hepatic enlargement should prompt caution in PD initiation. PD was selected based on the patient’s underlying condition and lifestyle preferences. In light of the updated guideline, more cautious evaluation of PD suitability is warranted in patients with significant organ enlargement.

Previous studies have shown that, compared with patients undergoing PD with other underlying diseases, those with ADPKD do not differ in dialysis dose (Kt/V), peritoneal leakage, peritonitis, transition to HD, technique failure, exit-site infection, or mortality; however, the incidence of abdominal hernia is higher in patients with ADPKD [20]. Increased IAP secondary to organ enlargement is considered one contributing factor, and PD may further elevate IAP; therefore, careful attention should be paid to dialysate volume.

The diagnosis of abdominal hernia is primarily clinical, and ultrasonography or CT is recommended when the diagnosis is unclear [21]. However, obturator hernia may present without specific clinical features or physical examination findings. Thus, imaging evaluation should be considered when suspicious findings are present. In addition, obturator hernias have been reported to be detected on the contralateral side in approximately 25% of cases during surgery [22]; therefore, evaluation of the contralateral obturator foramen may be useful at the time of operation. In this case, the contralateral side was not evaluated during the initial surgery, and such assessment might have reduced the risk of recurrence.

Although most patients can resume PD by postoperative day 3, gradual increases in fill volume based on the time elapsed since abdominal surgery are recommended [21]. In the present case, PD was resumed 19 days after surgery; nevertheless, hernia recurrence occurred, necessitating a switch to HD. Individualized decisions regarding the timing and manner of PD resumption, considering patient-related and surgical factors, may help reduce the risk of hernia recurrence.

In summary, this is the first reported case of recurrent obturator hernia in a patient with ADPKD undergoing PD. Patients with ADPKD undergoing PD have a high incidence of abdominal hernias, and careful attention is required during PD initiation, maintenance, and management after hernia occurrence. Obturator hernias have a high morbidity and mortality rate, making it important for clinicians to consider this diagnosis in the presence of symptoms such as bowel obstruction or abdominal pain.

Author contributions

Conceptualization, visualization, writing—original draft, review, editing: KO; writing—review, editing: TK, YA, HT, NS. Each author contributed important intellectual content during manuscript drafting or revision and agrees to be personally accountable for their own contributions and to ensure that questions pertaining to the accuracy or integrity of any portion of the work—even one in which the author was not directly involved—are appropriately investigated and resolved, including with documentation in the literature if appropriate.

Funding

No funding was received to assist with the preparation of this manuscript.

Data availability

The data that support the findings of this case report are not publicly available due to privacy and ethical restrictions.

Declarations

Ethical approval

According to our institutional policy, ethical approval was not required for single case reports.

Informed consent

Informed consent to publish the details of the case was obtained from the patient.

Conflict of interests

The authors have no competing interests to declare that are relevant to the content of this article.