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| CASE REPORTS |
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| Year : 2022 | Volume
: 5
| Issue : 2 | Page : 83-85 |
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Hernia uterine inguinale in adolescence: A rare presentation
Angeline Mary Samy, Dasarathan Shanmugam
Department of General Surgery, JIPMER, Puducherry, India
| Date of Submission | 21-May-2021 |
| Date of Decision | 31-May-2021 |
| Date of Acceptance | 16-Jun-2021 |
| Date of Web Publication | 19-May-2022 |
Correspondence Address:
Dr. Angeline Mary Samy
Department of General Surgery, JIPMER, Puducherry
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/ijawhs.ijawhs_31_21
A 14-year-old girl presented with a swelling over the right groin for 2 years associated with amenorrhea. She also had cyclical pain over the swelling every month. On further evaluation, she had a right inguinal hernia with right ovary and uterus in the hernia sac. She underwent inguinal exploration and hernial sac was identified. She underwent hernioplasty and was discharged uneventfully. The presence of uterus in a hernial sac is a rare entity. The risk of complications and chances of untoward injury during the unprecedented surgical procedure warrants a careful evaluation before surgery. Keywords: Hernial sac, inguinal exploration, inguinal hernia, meshplasty
How to cite this article:
Samy AM, Shanmugam D. Hernia uterine inguinale in adolescence: A rare presentation. Int J Abdom Wall Hernia Surg 2022;5:83-5 |
| Introduction | |  |
Hernia uterine inguinale (HUI) is a rare condition usually seen in the pediatric population.[1] It may be associated with other congenital anomalies, especially urogenital developmental anomalies.[2] The processus vaginalis in boys corresponds to the diverticulum of Nuck in girls.[3] It is a peritoneal outpouching that extends from the uterus to the labia majora. A widely patent processus vaginalis may permit herniation of abdominal contents. It is very unusual to have uterus and adnexa in the hernial sac. We observed an adolescent girl with atypical presentation of swelling in the right inguinal region with cyclical pain which raised our suspicion and, on further evaluation, observed a rare presentation of an inguinal hernia.
| Case Presentation | | |
A 14-year-old girl presented with a swelling in the right groin for 2 years associated with cyclical pain over the swelling. The swelling increases in size every month correspondingly. On examination, she was of short stature and had multiple congenital anomalies as shown in [Figure 1]A–C. A 4 cm × 3 cm firm, non-tender, irreducible swelling in the right inguinal region with expansile cough impulse was noted. In view of multiple congenital anomalies, she was enquired retrospectively. She revealed that her mother had taken an unknown abortifacient during the second trimester of pregnancy. Ultrasonogram revealed a 17 mm defect in the right lower anterior abdominal wall with herniation of uterus and right ovary with minimal free fluid in the hernial sac. Her ECHO revealed mild mitral regurgitation and bicuspid aortic valve. Magnetic resonance imaging showed the presence of uterus, right ovary, and fluid filled hernia sac in the subcutaneous plane with a defect in the inguinal wall, as shown in [Figure 2]. | Figure 1: (A) Congenital anomaly of high arched palate seen in this photograph. (B) Photograph showing ectrodactyly of right hand. (C) Photograph showing low set ears
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 | Figure 2: MRI abdomen—T2 image showing uterus and right ovary with free fluid within the right inguinal hernial sac
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She underwent right inguinal exploration. Intraoperatively, indirect inguinal hernia was noted with uterus, right ovary, and right fimbria as contents in the hernial sac. Dense adhesions were present between uterus, ovary, and hernial sac, as shown in [Figure 3] and [Figure 4]. Ligamentum rotundum was not visualized as a part of hernial sac contents. Adhesions were gently released and contents were reduced. The size of the defect was found to be 2.5 cm × 2 cm. The deep inguinal ring was closed with additional 2-0 vicryl sutures, and the defect was obliterated. Inguinal hernioplasty was accomplished using a prolene mesh. A 3″×6″ (inch) large pore prolene mesh was placed in the space below external oblique aponeurosis and fixed using 2-0 prolene sutures. The total duration of the surgery was 1 h and 30 min. | Figure 3: Intraoperatively, uterus, right ovary with fimbria seen within the hernial sac
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 | Figure 4: Intraoperatively, adhesions noted between uterus, fimbria, and right ovary within the hernial sac
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The patient had an uneventful recovery in the post-operative period. She was started on orals on the same day and escalated to normal diet on next day. She was discharged on post-operative day 3. Her wound was healthy. The patient was followed up for a period of 1 year. There was no evidence of recurrence.
| Discussion | | |
There have been very few reported instances of the presence of uterus in the hernia sac. Akıllıoğlu et al.[4] reviewed 3100 female children with inguinal hernia and reported the incidence of HUI of 0.23% with a mean age of 7.4 months and it decreases with age. It was usually seen in neonates with associated karyotype anomalies. It was commonly observed in hermaphrodites, intersexual variants, and male pseudohermaphroditism. In female phenotype, uterus was a part of sliding hernia and extremely rare.[4] The presence of uterus in a hernial sac in adolescence is very rare. The most common presentation of HUI was a groin or labial swelling. Vaginal bleeding is an uncommon presentation as reported by Zitsman et al.[5] In adult women with HUI, Kokcu et al.[6] observed genital abnormalities such as bicornuate uterus, ipsilateral uterine horn, ovarian ligament, Fallopian tube More Details, and ovary in the hernial sac and usually presented with infertility. However, in our case, there were no genital abnormalities and her karyotype was 46XX. She had developmental anomalies of short stature, high arched palate, low set ears with limb anomalies, which was found to be an odd presentation and raises the suspicion of a syndromic association. Canal of Nuck is more commonly open in premature infants, which may lead to herniation as proposed by Chavan.[7] In our case report, the patient had a normal term delivery. In a retrospective analysis of 1,084 female babies with inguinal hernia by Dreuning et al.,[8] hernial sac contained ovary in 21.7% of the patients, and ectopic testis was found in 0.6% of the patients. Only three cases of indirect hernia and one case of direct hernia containing whole or part of the uterus, fallopian tubes, and ovaries have been described in literature. The presence of uterus with one ovary has been described in six case reports.[3]
The reasons proposed for HUI are elongated uterine suspensory ligaments. Primary weaknesses of the uterine and ovarian suspensory ligaments may also cause herniation of uterus or adnexa.[3] These anatomic abnormalities might occur due to hormonal disorders because of medications used during pregnancy in artificial reproductive techniques. Another reason proposed was the failure of fusion of Mullerian ducts causing excessive mobility of the ovaries and non-fusion of uterine cornuae.[9] This could explain the increased incidence in premature babies and disorders of sexual development (DSD) in cases of anti-Müllerian hormone insensitivity, persistence of Müllerian derivatives, and ovotestis DSD.
| Learning Points/Take Home Messages | | |
Various anatomic anomalies may compromise fertility; therefore, careful physical evaluation is essential. Ultrasound may be an initial diagnostic tool in screening any suspicious clinical findings.
Various surgical options to be explained to the patient and counsel regarding the surgery and its sequelae.
A tailored approach is necessary to avoid embarrassment intraoperatively and to reduce the risk of unprecedented injuries.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient and the mother have given their consent for her images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Karadeniz Cerit K, Ergelen R, Colak E, Dagli TE Inguinal hernia containing uterus, fallopian tube, and ovary in a premature newborn. Case Rep Pediatr 2015;2015:807309.  |
| 2. | Bar-Joseph KL, Amies-Oelschlager AM, Avansino JR A painful protuberance: A young woman had primary amenorrhea and an inguinal hernia. Am J Obstet Gynecol 2012;207:144.e1-2. |
| 3. | Cascini V, Lisi G, Di Renzo D, Pappalepore N, Lelli Chiesa P Irreducible indirect inguinal hernia containing uterus and bilateral adnexa in a premature female infant: Report of an exceptional case and review of the literature. J Pediatr Surg 2013;48:e17-9. |
| 4. | Akıllıoğlu I, Kaymakcı A, Akkoyun I, Güven S, Yücesan Ş, Hiçsönmez A Inguinal hernias containing the uterus: A case series of 7 female children. J Pediatr Surg 2013;48:2157-9. |
| 5. | Zitsman JL, Cirincione E, Margossian H Vaginal bleeding in an infant secondary to sliding inguinal hernia. Obstet Gynecol 1997;89:840-2. |
| 6. | Kokcu A, Malazgirt Z, Cetinkaya MB, Tosun M Presence of a uterine horn and fallopian tube within an indirect hernial sac: Report of a rare case. Hernia 2010;14:325-7. |
| 7. | Chavan. Hernia uterine inguinale [Internet]. Available from: http://www.saudisurgj.org/article.asp?issn=2320–3846;year=2016;volume=4;issue=2;spage=84;epage=86;aulast=Chavan. [Last accessed on 2020 May 5]. |
| 8. | Dreuning KM, Barendsen RW, van Trotsenburg AP, Twisk JW, Sleeboom C, van Heurn LE, et al. Inguinal hernia in girls: A retrospective analysis of over 1000 patients. J Pediatr Surg2020;55:1908-13. Available from: http://www.sciencedirect.com/science/article/pii/S0022346820302396. [Last accessed on 2020 May 5]. |
| 9. | Ming YC, Luo CC, Chao HC, Chu SM Inguinal hernia containing uterus and uterine adnexa in female infants: Report of two cases. Pediatr Neonatol 2011;52:103-5. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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