Ovarian Incarceration and Torsion in Single-Ovary Versus Multiple-Reproductive Organ Prolapse in Female Inguinal Hernia: A Retrospective Study of 510 Infants Who Underwent Laparoscopic Hernia Repair

Abstract

Purpose:

Although inguinal hernia repair is common in infants, few studies have focused on the condition in female infants. In female infantile inguinal hernia, there is a risk of oophorectomy when torsion and strangulation occur due to ovarian prolapse. We aimed to evaluate the risks of ovarian incarceration and torsion in single-ovary versus multiple-reproductive organ prolapse in female infantile inguinal hernia.

Methods:

Females ≤12 months old who underwent laparoscopic transabdominal inguinal hernia repair from September 2012 to December 2019 were retrospectively analyzed. If manual reduction failed at initial diagnosis, surgery was performed within 24 hours in all incarceration cases. The clinical characteristics and surgical outcomes were compared between those with single-organ versus multiple-organ prolapse.

Results:

Of 510 patients, 465 (91.2%) had single-organ prolapse, most commonly a single ovary (381/465), followed by intestine (84/465). Forty-five patients (8.8%) had multiple-organ prolapse, most commonly a single ovary plus intestine (27/45), followed by both ovaries plus the uterus (10/45). The manually irreducible incarceration rate was higher in patients with multiple-organ prolapse (57.8%) than single-organ prolapse (23.4%; P < .000). In patients with ovarian incarceration, the ovarian torsion rate was higher in the single-ovary group (38/109, 34.9%) than the multiple-organ group (1/26, 3.8%; P < .000). There was no significant difference between the two groups in recurrence or oophorectomy rate.

Conclusion:

Most female infantile inguinal hernias involve ovarian prolapse. There is a high risk of incarceration in multiple-organ prolapse and a high risk of ovarian torsion in single-ovary prolapse.

Introduction

Indirect inguinal hernia repair is one of the most common procedures performed by pediatric surgeons. The overall prevalence of indirect inguinal hernia ranges from 0.8% to 5% in full-term infants.¹ Inguinal hernias are more common in boys than in girls (5:1 ratio).² An abnormal open pouch of the peritoneum extending into the female labia majora was described by Anton Nuck in 1691 and is referred to as the canal of Nuck. The canal of Nuck is a homolog of the processus vaginalis in males and can result in inguinal hernia in females.

The inguinal hernial sac often contains intestine and omentum in boys and ovary and uterus in girls.³ In female infants, the inguinal hernia commonly contains an ovary, which is often incarcerated.⁴ Hernias in female infants may also contain various intra-abdominal organs, including the uterus.^5,6 The rate of organ incarceration is higher in patients younger than 1 year than in older patients.⁷ Incarceration in boys mainly involves a single organ (usually the intestine or omentum), whereas girls can have incarceration of multiple organs into the canal of Nuck, including the ovary, uterus, intestine, and omentum.³ Special precautions are required during inguinal hernia repair in girls to avoid injuring incarcerated reproductive organs.^8,9 Incarcerated ovaries can undergo torsion, which increases the risk of strangulation.^10,11

Most comprehensive studies of pediatric inguinal hernia have included both boys and girls; few have evaluated the characteristics of inguinal hernia in a large population of female infants. Only a few case series have been reported, usually involving complicated multiple-organ incarceration, including the uterus and ovary. Continued ovarian torsion increases the risk of oophorectomy, and incarceration increases the risk of strangulation. Therefore, the aims of the present study were to identify the characteristics of inguinal hernias in female infants and to evaluate the risk of ovarian incarceration and torsion in single-ovary versus multiple-reproductive organ prolapse in a relatively large cohort of female infants.

Materials and Methods

The medical records of female infants younger than 1 year of age who had undergone laparoscopic inguinal hernia repair from September 2012 to December 2019 in the surgical department specializing in hernia repair at Damsoyu Hospital, Seoul, Korea were retrospectively analyzed. The following data were collected from the medical records: patient age at surgery; pre-term and low birth weight status; hernia laterality, contents, and reducibility; interval from onset of symptoms to surgery; duration of surgery; presence of ovarian torsion; organ resection; postoperative complications; and recurrence.

All surgeries were performed by a highly experienced surgeon with 15 years of laparoscopic experience (S.R. Lee). Eleven patients with confirmed androgen insensitivity syndrome were excluded from the analysis. The prolapsing organs were typically diagnosed with ultrasonography, with findings similar to those reported by Jedrzejewski et al.¹² The ultrasonographic findings included either a hypoechoic oval structure (ovary and fallopian tube, Fig. 1a) or multiple hyperechoic tubular structures (intestine, Fig. 1b). However, with multiple-organ incarceration, it was difficult to distinguish organs with diagnostic ultrasonography (Fig. 1c, d); 45 patients with two or more prolapsed organs were diagnosed during laparoscopic surgery.

FIG. 1.

Ultrasonographic and laparoscopic findings in female pediatric patients with inguinal hernias. (a) Single ovary. (b) Intestine. (c) Both ovaries and the uterus. (d) Intestine and ovary.

Manual reduction was initially attempted, and laparoscopic intracorporeal reduction was performed if manual reduction failed. If manual reduction failed at initial diagnosis, surgery was performed within 24 hours in all cases. During surgery, the organ prolapsed through the deep inguinal ring was confirmed with a laparoscopic camera. If the prolapsed organ was reduced by external compression, it was not considered incarcerated.

Patients were categorized into the multiple- or single-organ prolapse group, and parameters were compared between these groups. Patients underwent surgery only after the procedure had been described to their caregiver(s), and the caregivers' written informed consent was obtained. This study was approved by the Institutional Review Board of Damsoyu Hospital (approval number DSY-2018-002). All procedures performed were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Laparoscopic inguinal hernia repair technique

The surgical procedures were performed with the patient in the supine position under general anesthesia. The laparoscopic system comprised a 2.9-mm camera with 2.7-mm instruments. A transumbilical incision was made with a no. 15 scalpel blade to allow insertion of a trocar, through which pneumoperitoneum was induced and maintained with carbon dioxide insufflation at 6–7 mmHg. Two other instruments were inserted through separate 3.0-mm stab incisions in the lateral abdomen.

Intracorporeal reduction was successful in most patients with a manually irreducible incarcerated hernia containing ovary. Moreover, intracorporeal reduction was performed without difficulty in patients with an inguinal hernia containing both ovaries and the uterus; the order of reduction in these patients was: contralateral ovary, uterus, and then ipsilateral ovary (Fig. 2).

FIG. 2.

Laparoscopic views of multiple-organ incarceration. (a) Intestinal (black arrow) and single-ovarian (white arrow) incarceration. (b) Single-ovarian (white arrow) and uterine incarceration. (c) Bilateral-ovarian (white arrow) and uterine (black arrow) incarceration. (d) Bilateral-ovarian, uterine (white arrow), and intestinal (black arrow) incarceration. Intracorporeal reduction of multiple incarcerated organs. (e) Simultaneous ovarian and uterine incarceration. (f) Intracorporeal reduction of the contralateral ovary (white arrow) is followed by (g) intracorporeal reduction of the uterus (white arrow) and (h) intracorporeal reduction of the ipsilateral ovary (white arrow).

In patients with an inguinal hernia containing intestine and a single ovary, intracorporeal reduction comprised reduction of the intestine and then the ovary. After removing the hernial sac, intracorporeal repair was performed using nonabsorbable, multifilament, 3.0–4.0 silk, as previously reported.^3,13 The contralateral canal of Nuck was then examined for patency, and the open canal was sutured if necessary.

Statistical analyses

All statistical analyses were performed with R software version 3.6.1 (R Development Core Team, Vienna, Austria). Continuous variables are presented as mean and standard deviation; categorical variables are presented as frequency and percentage. The Shapiro–Wilk test was used to test for normality of continuous variables, and the t-test or Wilcoxon's rank sum test was used for continuous variables. Fisher's exact test or the χ² test was used for categorical variables. The significance threshold was set at P < .05; any univariate P value ≤.05 was considered statistically significant.

Results

Patient characteristics and outcomes

Table 1 shows the demographic characteristics of the 510 patients. All patients underwent successful laparoscopic hernia repair, and none required conversion to open surgery. Unilateral hernia (465/510, 91.2%) was much more common than bilateral hernias (45/510, 8.8%). Among unilateral hernias, 56% (262/465) were left sided. Of the 510 patients, 465 (91.2%) had single-organ prolapse, most commonly a single ovary (381/465), followed by intestine (84/465). Forty-five of the 510 patients (8.8%) had multiple-organ prolapse, most commonly a single ovary plus intestine (27/45), followed by both ovaries plus the uterus (10/45). All hernias containing multiple organs included at least one ovary (Fig. 2).

Table 1.

Demographic Characteristics, Hernia Characteristics, and Surgical Outcomes of Female Infants with Inguinal Hernia

	Total (n = 510)
Age (months)	4.1 ± 3.7 (0.7–12.0)
Pre-term birth (<37 weeks)	80 (15.7%)
Low birth weight (<2.5 kg)	77 (15.1%)
Hernia location
Right	203 (39.8%)
Left	262 (51.4%)
Bilateral	45 (8.8%)
Prolapsed organ(s)
Single organ (n = 465)
Intestine	84 (16.5%)
Ovary	381 (74.7%)
Multiple organs (n = 45)
Single ovary and the uterus	7 (1.3%)
Both ovaries and the uterus	10 (2.0%)
Single ovary and intestine	27 (5.3%)
Both ovaries, uterus, and intestine	1 (0.2%)
Manually irreducible incarceration (laparoscopic intracorporeal reduction)	135 (26.5%)
Organ resection
Oophorectomy	1 (0.2%)
Postoperative complications
Hematoma	2 (0.4%)
Seroma	0 (0.0%)
Wound infection	1 (0.2%)
Metachronous contralateral inguinal hernia	0 (0.0%)
Recurrence	2 (0.4%)
Follow-up period (months)	49.3 ± 24.6 (6–93)

Categorical variables are presented as number (%); continuous variables are presented as mean ± standard deviation (range).

Patients are included only once in the percentage denominators (i.e., patients with bilateral symptoms are counted as a single patient).

Oophorectomy was performed in one patient with an unrecovered ischemic ovary in the single-ovary incarceration group. Postoperative complications included two inguinal hematomas and one umbilical wound infection. Two recurrences were observed during the follow-up period. Recurrence occurred in one patient in the multiple organ group and one patient in the single organ group; both were incarcerated hernias.

Comparison of the single- and multiple-organ prolapse groups

Table 2 shows the clinical characteristics of patients with multiple- versus single-organ prolapse. The mean duration from onset of symptoms to surgery did not significantly differ between the single-organ (17.5 days) and multiple-organ (19.2 days) hernia groups. The prevalence of manually irreducible incarceration was significantly higher in the multiple-organ group than in the single-organ group (57.8% [26/45] vs. 23.4% [109/465]; P < .000). The operation time was longer in the multiple-organ group than in the single-organ group (20.5 ± 8.7 minutes [range, 9–45 minutes] vs. 17.2 ± 8.0 minutes [range, 8–50 minutes]; P = .008). The recurrence rate did not significantly differ between the two groups.

Table 2.

Comparison of Clinical Characteristics Between Patients with Multiple-Versus Single-Organ Prolapse in Inguinal Hernia

	Multiple organs (n = 45)	Single organ (n = 465)	P-value
Age (months)	3.0 ± 2.8 (1.0–9.0)	4.3 ± 3.8 (0.7–12.0)	.059
Weight (kg)	5.8 ± 3.5 (3.4–26.0)	6.1 ± 2.0 (2.8–11.0)	.048
Pre-term birth (<37 weeks)	6 (13.3%)	74 (15.9%)	<.000
Low birth weight (<2.5 kg)	9 (20.0%)	68 (14.6%)	<.000
Laterality			.468
Right	17 (37.8%)	186 (40.0%)
Left	26 (57.8%)	236 (50.8%)
Bilateral	2 (4.4%)	43 (9.2%)
Duration from onset of symptoms to surgery (days)	17.5 ± 29.1 (1.0–61.0)	19.2 ± 32.5 (0.0–180.0)	.565
Manually irreducible incarceration (laparoscopic intracorporeal reduction)	26 (57.8%)	109 (23.4%)	<.000
Operation time (min)	20.9 ± 8.7 (9.0–45.0)	17.6 ± 8.0 (8.0–50.0)	.008
Recurrence	1 (2.2%)	1 (0.2%)	1.000

Categorical variables are presented as number (%); continuous variables are presented as mean ± standard deviation (range).

Continuous variables were tested with the Wilcoxon rank-sum test.

Categorical variables were tested with the χ² test or Fisher's exact test.

Comparison of the single-ovary and multiple-organ incarceration groups

Table 3 summarizes the characteristics of patients with ovarian incarceration. Among the patients with ovarian incarceration, the mean duration of symptoms did not differ between the multiple-organ group (21.0 days) and the single-ovary group (9.6 days). The ovarian torsion rate in the multiple-organ incarceration group (3.8%, 1/26) was significantly lower than that in the single-ovary incarceration group (34.9%, 38/109; P < .000). The prevalence of ovarian strangulation did not differ between the multiple-organ group (11/26, 42.3%) and the single-ovary group (55/109, 50.5%). Detorsion of the ovary was performed for incarcerated hernias characterized by ovarian torsion (Fig. 3). Color change in the organs was evaluated after high ligation, and organ viability was confirmed in most patients. Oophorectomy was performed in one patient in whom ovarian ischemia did not resolve after successful reduction.

FIG. 3.

Ovarian torsion in incarcerated inguinal hernia. (a) Incarceration and torsion (white arrow) of the left ovary. (b) Intracorporeal reduction of the incarcerated ovary with torsion (white arrow). (c) Torsion release (detorsion, white arrow). (d) Incarceration of the left ovary. (e) Intracorporeal reduction of the incarcerated ovary with torsion (white arrow). (f) Torsion release (detorsion, white arrow).

Table 3.

Comparison of Surgical Outcomes Between Patients with Incarcerated Hernia Contents Containing Multiple Organs Versus a Single Ovary

	Multiple-organ incarceration (n = 26)	Single-ovary incarceration (n = 109)	P-value
Age (months)	3.5 ± 3.0 (1.0–9.0)	3.1 ± 3.1 (0.7–12.0)	.235
Weight (kg)	5.8 ± 1.7 (3.9–9.0)	5.5 ± 1.7 (3.0–10.0)	.261
Pre-term birth (<37 weeks)	3 (11.5%)	17 (15.6%)	.002
Low birth weight (<2.5 kg)	4 (15.4%)	13 (11.9%)	.029
Laterality			.281
Right	12 (46.2%)	41 (37.6%)
Left	14 (53.8%)	59 (54.1%)
Bilateral	0 (0.0%)	9 (8.3%)
Duration of symptoms (days)	21.0 ± 32.3 (1.0–61.0)	9.6 ± 29.1 (1–50)	.535
Ovarian torsion	1 (3.8%)	38 (34.9%)	<.000
Ovarian strangulation	11 (42.3%)	55 (50.5%)	.597
Oophorectomy	0 (0.0%)	1 (1.0%)	1.000

Categorical variables are presented as number (%); continuous variables are presented as mean ± standard deviation (range).

Continuous variables were tested with the Wilcoxon rank-sum test.

Categorical variables were tested with the χ² test or Fisher's exact test.

Discussion

In this study of 510 female infants with inguinal hernia, ovarian prolapse was present in all patients with multiple-organ prolapse and in 74.7% of those with single-organ prolapse. This finding agrees with the study of Merriman and Auldist, in which 82% of incarcerated hernias in female patients contained ovaries.¹¹ Prolapse of the uterus and uterine adnexa through an inguinal hernia has rarely been reported in female pediatric patients.^6,14 Prolapse of the gynecologic organs is age related, with 70% of affected patients younger than 5 years of age.^15,16 No large cohort study of multiple-organ prolapses or comparison of incarceration risk between single-ovary and multiple-organ prolapse has been reported.

In the present study, incarceration of hernia contents was significantly more common in patients with multiple-organ prolapse than those with single-organ prolapse. Among 109 patients with single-ovary incarceration, eight patients showed a severely ischemic ovary in the hernial sac. Seven of these eight patients showed improvement of ischemia after intracorporeal reduction and torsion release. After verifying congestion relief with improved blood flow, the ovaries were able to be preserved in seven of these patients. However, one patient required oophorectomy because there was no improvement in blood flow even after torsion release. These results conflict with those of Merriman and Auldist, who reported that 5 of 71 patients with ovarian incarceration required oophorectomy.¹¹

Incarcerated ovaries can undergo torsion, leading to lymphatic and venous obstruction, which increases the risk of strangulation. Prolonged incarceration causes arterial obstruction and increases the risk of requiring oophorectomy.^17,18

The incarceration rate was higher in the multiple-organ group than in the single-organ group in the present study. However, ovarian torsion was more common in the single-organ group than in the multiple-organ group. Two presumptions may be made on the basis of these findings. The first is that multiple-organ prolapse was associated with a higher incarceration rate due to pressure effects caused by large hernia volume, which can also lead to difficulty in reduction. The second presumption is that the risk of ovarian torsion was probably higher in the single-organ group because these patients had a relatively larger space in the canal of Nuck, which enabled the prolapsed ovary to move and rotate in the hernial space.

In the present study, the mean duration of symptoms was 17 days in the multiple-organ group, but was 21 days in the incarcerated subgroup. In contrast, the mean duration of symptoms was 15 days in the single-organ group, but was 9 days in the incarcerated subgroup. In the present cohort, oophorectomy was successfully avoided in all but one patient due to the performance of all surgeries within 24 hours of the diagnosis of an incarcerated hernia. In cases with ovarian edema in an incarcerated hernial sac, the most effective way to reduce the incarcerated edematous organs seemed to be intracorporeal pulling of the incarcerated organs into the abdominal cavity individually rather than external compression. Other case reports also highlight the risk of incarceration in patients with multiple-organ prolapse.^14,19

In the present study, the operation time was longer in the multiple-organ group than in the single-organ group. However, the average operation time was 20 minutes in the multiple-organ group, which is very short compared with the 53 minutes for laparoscopic infantile ovarian hernia repair reported by Saeki et al.²⁰ Although many variables contribute to surgery time, the most important factor may be surgical experience.

The mean duration of symptoms in infantile hernial incarceration was only 21 days in the multiple-organ group and 9.6 days in the single-organ group. Although the mean duration of symptoms did not significantly differ between the single- and multiple-organ groups, the period between the onset of hernia symptoms and incarceration tended to be longer in the multiple-organ group than in the single-organ group. As the infant females were too young to communicate directly, the onset of symptoms was set as the time at which the parents first noticed inguinal bulging. As the onset of hernia symptoms is very difficult to determine in pediatric patients, it is not possible to accurately make recommendations regarding the timing of surgery based on the onset of symptoms. However, it is very important to consult a surgeon as soon as possible for infantile female inguinal hernia, as incarceration may occur within 1 day after the onset of hernia symptoms in both groups.

Other studies have reported that prompt surgery is required for inguinal hernias in female infants younger than 1 year.^6,21 The rate of oophorectomy at our institution (0.7%, 1/135 incarcerated hernias [26 multiple-organ and 109 single-ovary prolapses]) was lower than previously reported data, most likely because the torsion was released immediately after intracorporeal reduction of the incarcerated ovary.²² Previous studies have reported an oophorectomy rate among patients with ovarian incarceration undergoing laparoscopic hernia repair of 33.3% (1/3)²² and 6.7% (1/15).¹⁸

In female infants with inguinal hernias, torsion can occur because of the pedunculated structure of the ovaries and fallopian tubes. In the present study, the ovarian torsion rate among patients with ovarian incarceration was 28.9% (39/135 [26 patients in the multiple-organ group and 109 in the single-ovary group]). These results conflict with previous studies that reported ovarian torsion rates of 19.0% (11/58)¹¹ and 0.0%.²³

Although the prevalence of ovarian torsion differs across studies, it is important to perform rapid reduction to preserve the incarcerated ovary. Laparoscopic intracorporeal reduction was possible in all of the present patients, and the rate of oophorectomy was markedly low. This low rate of oophorectomy supports the findings of a previous study that reported that severely ischemic torsed ovaries were successfully preserved after detorsion with or without hematoma evacuation.²⁴ However, it is ideal to treat inguinal hernia before ovarian torsion occurs.

Some limitations of the present study should be noted. Because of the relatively low prevalence of inguinal hernia in female infants, the study was retrospective in design. However, the study population was relatively large, which increases the significance of the findings. This was a single-center study and thus the findings may not be generalizable to other institutions and populations. Future multicenter studies are needed to confirm the present findings. Long-term studies are needed to evaluate the effects of ovarian torsion and incarceration on future fertility.

Conclusions

In this large single-center study, most inguinal hernias in female infants contained a prolapsed ovary. Female infants with multiple-organ prolapse had a high risk of incarceration, and those with single-ovary prolapse had a high risk of ovarian torsion. Laparoscopic hernia repair was effective in the treatment of ovarian torsion and incarceration.

Footnotes

Acknowledgments

The author thanks Beom Seok Oh and Young Hyun Lee from the Research Center of Damsoyu Hospital for assisting with the statistical analysis and organizing the data in this article. The author also thanks Kelly Zammit, BVSc, Angela Morben, DVM, ELS, Jane Charbonneau, DVM, and Rebecca Tollefson, DVM, from Edanz Group, for editing drafts of this article.

Disclosure Statement

Sung Ryul Lee has no conflict of interest or financial ties to disclose.

Ethical Approval

This study was approved by the Institutional Review Board of Damsoyu Hospital (approval number DSY-2018-002).

Human and Animal Rights

All procedures performed were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed Consent

Informed consent was obtained from the guardians of all individual participants included in the study.

Funding Information

No funding was received for this article.

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