Complete uterine and bilateral adnexal herniation in a female infant: a late-presenting case report

This case demonstrates that complex reproductive organ herniation, including the uterus and bilateral adnexa, can occur in reducible hernias and can present beyond early infancy, even in the absence of alarming symptoms. A high index of suspicion is necessary, and routine preoperative ultrasonography should be strongly considered for all female infants with inguinal hernias to ensure accurate diagnosis and optimal surgical management focused on fertility preservation.

Indirect inguinal hernia represents one of the most common congenital anomalies in infancy, with an incidence ranging from 0.8% to 4.4% in the general pediatric population and rising to 30% among preterm neonates[1,2]. While boys are disproportionately affected (ratio 6:1), hernias in female infants carry distinct clinical significance due to the potential involvement of reproductive organs. The ovary and fallopian tube are documented in 15–20% of female cases, often presenting as sliding components within the hernia sac[1–3]. In stark contrast, herniation of the uterus alongside the adnexa – termed hernia uteri inguinalis – is exceptionally rare in phenotypically and genotypically normal female infants[1,2,4]. Current literature comprises predominantly isolated case reports or small series, with only 73 pediatric cases systematically reviewed to date[2]. This anomaly typically manifests as an asymptomatic reducible mass or, less commonly, an incarcerated hernia requiring urgent intervention[2,5]. The pathophysiology remains incompletely elucidated but may involve abnormal mobility of uterine ligaments or delayed closure of the canal of Nuck[3,4]. Diagnostic precision is critical, as the presence of reproductive organs elevates risks of incarceration, vascular compromise, and iatrogenic injury during surgery[1,5]. High-resolution ultrasonography (HRUS) with Doppler is the cornerstone for preoperative identification of uterine/adnexal structures, enabling tailored surgical planning[1,2,4]. Despite consensus on early repair to prevent complications, debates persist regarding optimal timing (elective vs. urgent) and the necessity of contralateral exploration[2]. Herein, we report a unique case of a 1-year-old female infant with an elective left inguinal hernia repair, revealing the uterus, bilateral ovaries, and fallopian tubes within the sac. To our knowledge, this represents one of the few documented cases involving complete bilateral adnexal herniation alongside the uterus in a non-incarcerated setting. This case underscores the importance of considering atypical contents in female infant hernias, even beyond the neonatal period. This case report has been reported in line with the SCARE checklist[6]. Case presentation A 1-year-old phenotypically normal Asian female infant presented to the surgery clinic at a tertiary care center. A history of intermittent, non-tender left inguinal swelling since early infancy was noted. Parents reported spontaneous reduction when supine or with gentle pressure. There were no signs of incarceration (vomiting, erythema) or systemic symptoms. There were no prior illnesses, hospitalizations, or surgeries. There were no inheritable disorders or hernia history reported. The infant has no regular medications or known drug allergies or adverse reactions documented. Systems review was unremarkable (no fever, weight loss, gastrointestinal, or genitourinary symptoms). Soft, reducible left inguinal mass (~2 cm) was notable on physical examination with no skin changes. It was non-tender, fully reducible. No cough impulse was exhibited. Imaging investigations were not performed, as the diagnosis of inguinal hernia in infants is primarily clinical. Prognosis remains favorable due to the reducible nature, absence of ischemia, and successful anatomical reduction. Preoperative optimization involved standard nil-by-mouth fasting with no medication adjustments required. Elective open left inguinal herniotomy was performed under general anesthesia with single-dose IV cefazolin prophylaxis. Annotated schematic of the hernia sac containing the herniated uterus and both ovaries, with ovarian arteries and veins, is illustrated (Fig. 1). Using the standard technique for symptomatic infant hernia repair, in supine position, an inguinal incision was made; sac contents were gently reduced; high ligation with 3-0 Vicryl absorbable suture was performed; and the inguinal canal was closed in layers without mesh. No novel techniques were employed. The procedure was completed by an experienced surgeon at a high-volume tertiary pediatric hospital. No deviations from the surgical plan occurred. Follow-up consisted of clinic reviews at 2 weeks and 6 months postoperatively, where clinical examination confirmed no recurrence of hernia and satisfactory wound healing. Due to the nature of the herniated reproductive organs, long-term annual gynecological review was recommended until puberty to monitor reproductive development, with no additional imaging or testing required during the documented follow-up period. Postoperatively, the patient demonstrated full adherence to wound care and activity restrictions. Outcomes included successful reduction/ligation with discharge within 24 hours, and at 6-month follow-up, there was no recurrence with a well-healed wound and parental satisfaction regarding functional recovery. No complications occurred (Clavien–Dindo Grade 0), attributable to prophylactic cefazolin administration, with zero morbidity or mortality recorded. Figure 1.: Annotated schematic illustration of the hernia sac containing the herniated uterus and both ovaries, with ovarian arteries and veins. The diagram clearly depicts the spatial relationships of the organs, providing a precise and instructive visualization of this rare clinical scenario. Courtesy to Takezoe T et al.

The presentation of an indirect inguinal hernia containing the uterus, ovaries, and fallopian tubes in a phenotypically normal female infant represents an extraordinary clinical entity, with our case adding to the sparse literature on this condition. This infant’s bilateral adnexal involvement alongside uterine herniation – observed during elective repair at 1 year of age – is exceptionally rare. Only 73 pediatric cases have been systematically documented to date, with bilateral ovarian involvement noted in merely 18.2% of cases[4]. The absence of incarceration or ischemic compromise in our patient contrasts with reports where 37.5% of cases presented with incarceration requiring urgent intervention[4]. This highlights the variable natural history of such hernias, where some remain reducible and asymptomatic beyond infancy. HIGHLIGHTS Exceptionally rare case of complete herniation of the uterus and bilateral adnexa (ovaries and fallopian tubes) within a reducible left inguinal hernia sac (hernia uteri inguinalis) in a phenotypically normal female infant. This unique case presented electively at 1 year of age with a spontaneously reducible, asymptomatic mass, lacking incarceration, torsion, or ischemia – a scarcely reported occurrence beyond the neonatal period. The omission of preoperative high-resolution Doppler ultrasound (HRUS) underscores its mandatory role in all female infants with inguinal masses to identify atypical contents like reproductive organs and assess viability. Intraoperative exploration unexpectedly revealed viable uterus and bilateral adnexa within the hernia sac, necessitating and achieving meticulous reduction and high ligation. Routine preoperative HRUS is essential for every female infant with an inguinal hernia to accurately delineate contents and guide fertility-preserving surgical planning. Elective repair is prudent upon diagnosis to prevent complications, and long-term gynecological follow-up is recommended to monitor reproductive development. The pathophysiology of uterine herniation remains incompletely elucidated. Persistent patency of the canal of Nuck – the female analog of the processus vaginalis – is a prerequisite, typically obliterated by 8 months’ gestation[1,7]. However, uterine involvement suggests additional anatomical factors. Proposed mechanisms include the following: (1) Elongated or lax uterine suspensory ligaments may permit uterine descent into the inguinal canal[2,7]. (2) The ovary, frequently the initial herniated organ, may pull the uterus via the utero-ovarian ligament[4]. 3) Müllerian duct anomalies; though partially excluded in our case (normal phenotype), incomplete Müllerian fusion (e.g., unicornuate uterus) is associated with uterine malposition and herniation risk[3,7]. The predominance of left-sided hernias (61% in Brooks et al’s review) further suggests asymmetric closure of the canal of Nuck or ligamentous development[4]. Our case underscores a critical dilemma: the omission of preoperative ultrasound. HRUS with Doppler is the diagnostic cornerstone, accurately identifying uterine tissue (hypoechoic structure with central endometrial stripe) and ovarian viability (vascular flow assessment)[1,5,7]. Ming et al emphasized that HRUS prevents misdiagnosis of atypical hernias and guides surgical planning[7]. In reducible masses, ultrasound sensitivity approaches 100%, enabling differentiation from hydroceles, lymphadenopathy, or neoplasms[1,4]. While clinical diagnosis suffices for simple hernias, our case reinforces that female infants with inguinal masses warrant routine ultrasonography to exclude reproductive organ involvement and assess incarceration risk[1,5]. The intraoperative finding of uterus and bilateral adnexa necessitated meticulous dissection to avoid iatrogenic injury to the fallopian tubes or ovarian vessels. Adhesions between organs and the hernia sac – reported in 21% of cases – increase technical complexity[5]. Key principles include: (1) gentle reduction, avoiding traction on the fallopian tubes to prevent vascular injury or future tubal occlusion[2]; (2) high ligation, the gold standard, though Brooks et al noted internal ring reinforcement in 23.6% of cases to prevent recurrence[4]; and (3) contralateral exploration, performed selectively (24.1% in Brooks et al), guided by clinical or sonographic suspicion[4,5]. Laparoscopic approaches (41.8% in recent series) offer direct visualization of adnexa, potentially reducing tubal injury risk compared to open techniques[2,4]. While incarceration mandates emergency surgery, the optimal timing for asymptomatic cases remains debated. Muta et al observed spontaneous uterine repositioning with growth in a premature infant, advocating delayed repair to facilitate safer surgery[3]. However, Brooks et al reported no regression in uterine-containing hernias, supporting elective repair once diagnosed to mitigate incarceration/torsion risks[2]. Long-term gynecological follow-up is prudent, as ligamentous laxity may predispose to ovarian torsion or fertility issues, though robust data are lacking[4,7]. This case exemplifies that female infant inguinal hernias may harbor unexpected reproductive organs, even bilaterally and beyond the neonatal period. Key lessons include: (1) high clinical suspicion, especially in premature infants or left-sided masses; (2) mandatory ultrasonography, for all female infants with inguinal hernias to define contents and viability; (3) individualized surgical planning, prioritizing adnexal preservation and considering laparoscopic visualization; (4) long-term monitoring, until puberty to assess reproductive organ development and function. Continued reporting of such cases will refine management paradigms for this rare but clinically significant entity. This exceptional case of a reducible left inguinal hernia containing the uterus and bilateral adnexa in a phenotypically normal 1-year-old infant underscores the potential for complex reproductive organ involvement even beyond the neonatal period. It reinforces the critical importance of preoperative ultrasonography in all female infants presenting with inguinal masses to accurately delineate hernia contents, assess viability, and guide meticulous surgical planning aimed at preserving fertility. Intraoperative vigilance is paramount to avoid iatrogenic injury to delicate structures. Elective repair remains prudent to mitigate risks of future incarceration or torsion. Long-term gynecological follow-up is recommended to monitor reproductive development, highlighting the need for sustained clinical awareness of this rare entity. Strengths This case report provides a rare documentation of complete bilateral adnexal herniation (uterus, ovaries, and fallopian tubes) in a reducible inguinal hernia diagnosed beyond the neonatal period. Its educational value is amplified by highlighting a critical practice gap: the omission of preoperative ultrasound despite established guidelines, thereby reinforcing the imperative for mandatory high-resolution imaging in all female infants with inguinal masses. The report adheres rigorously to SCARE guidelines, enhancing methodological transparency, and delivers actionable clinical recommendations supported by documented successful outcomes at 6-month follow-up.

Key limitations include the absence of preoperative ultrasound, which precluded objective confirmation of hernia contents and viability assessment before surgery, representing a missed diagnostic opportunity. Follow-up was limited to 6 months, insufficient to evaluate long-term reproductive outcomes (e.g., ovarian function or fertility), and no contralateral groin assessment was performed despite the bilateral nature of herniated adnexa. The evaluation did not include pelvic MRI or genetic testing to definitively exclude Müllerian anomalies, and findings reflect a single tertiary center’s experience, potentially limiting generalizability to low-resource settings. Ethical approval Ethics clearance was not necessary since the University (Damascus University) waives ethics approval for publication of case reports involving no patients’ images, and the case report is not containing any personal information. The ethical approval is obligatory for research that involves human or animal experiments. Consent Written informed consent was obtained from infant patient’s legal guardian for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. Research registration unique identifying number (UIN) Not applicable. Guarantor Ahmad Alkheder. Provenance and peer review Not commissioned; externally peer reviewed. Data availability statement No data are associated with this article.

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[1]. Kummari S, Subburam S, Chokkalingam SR. An indirect inguinal hernia in a neonate containing the uterus, ovary, and fallopian tube: a report of a rare case and a literature review. Cureus 2024;16:e65440. [3]. Muta Y, Odaka A, Inoue S, et al. Neonatal inguinal hernias containing the uterus: a case report on changes in uterine position. J Surg Case Rep 2023;2023:rjad503. [4]. Ming YC, Luo CC, Chao HC, et al. Inguinal hernia containing uterus and uterine adnexa in female infants: report of two cases. Pediatr Neonatol 2011;52:103–05. [5]. Karadeniz Cerit K, Ergelen R, Colak E, et al. Inguinal hernia containing uterus, fallopian tube, and ovary in a premature newborn. Case Rep Pediatr 2015;2015:807309. [6]. Kerwan A, Al-Jabir A, Mathew G, et al. Revised Surgical CAse REport (SCARE) guideline: an update for the age of Artificial Intelligence. Prem J Sci 2025;10:100079. [7]. Ueda J, Yoshida H, Makino H, et al. Right inguinal hernia encompassing the uterus, right ovary and fallopian tube in an elderly female: case report. J Nippon Med Sch 2016;83:93–96. Thought you might appreciate this item(s) I saw in International Journal of Surgery Case Reports. Your message has been successfully sent to your colleague. Some error has occurred while processing your request. Please try after some time. Export to End Note Procite

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