Spectacular finding during emergency caesarean section in a patient with long-term infertility – case report

Introduction

Infertility has been defined as the inability of couples of childbearing age to achieve pregnancy after one year of unprotected sexual intercourse⁽¹⁾. It has been estimated to affect between 8 and 12 percent of couples of reproductive age worldwide, and it could be of male, female or mixed cause⁽²⁾. For women, fertility declines between the ages of 25 and 30 years old, when we are talking about a healthy woman with no other associated conditions; in the presence of pathologies, fertility disorders could appear much earlier⁽³⁾. In about 15% of infertility cases, the cause is unknown⁽⁴⁾.

Infertility is a condition that can cause psychological, physical, mental, spiritual or medical damage, affecting both the patient and her partner⁽⁵⁾. Studies in recent years in the literature have highlighted the benefits of acupuncture, Chinese herbs, psychotherapy sessions, electrical stimulation, homeopathy, and hyperbaric oxygen therapy⁽⁶⁾.

In the case of female infertility, the causes described were multiple. Endometrial polyps, adenomyosis and uterine leiomyomas were the most common pathologies that could be easily discovered in women of childbearing age and could be the cause of infertility⁽⁷⁾. Polypectomy in the case of endometrial polyps, myomectomy, when possible, in the case of fibromatous uterus, could significantly increase the chances of achieving a spontaneous pregnancy⁽⁸⁾.

There was a study in the literature conducted in Romania at a public hospital, including 51 patients diagnosed with infertility, evaluated by well-established methods and treated hysteroscopically. It was found that most of these patients had a diagnosis of secondary infertility, and the treatment of endometrial causes by hysteroscopy significantly improved the chances of subsequent conception⁽⁹⁾.

Age was an important factor determining success in fertility treatments, and anti-Müllerian hormone (AMH) was a useful marker, showing ovarian reserve in women of reproductive age⁽¹⁰⁾. In the area of assisted reproductive technology, serum AMH levels are used to correlate the prognosis of ovarian stimulation, embryo quality, or pregnancy rates⁽¹¹⁾.

Müllerian duct anomalies were congenital malformations with an increased incidence, ranging from 0.5% to 6.7% in the general population⁽¹²⁾. The most commonly associated signs and symptoms described were primary amenorrhea, dysmenorrhea, pelvic pain, endometriosis, as well as problems related to sex life, with a significant impact on affected women⁽¹³⁾. In the case of uterine malformations, it has been discussed in the literature that they negatively influence fertility⁽¹⁴⁾; the most common was septate uterus in about 6-16% of cases⁽¹⁵⁾. Infertility or early spontaneous abortion when a patient has been diagnosed with uterine malformation has been associated with inadequate implantation of the embryo or poor vascularity of the septum, unfavorable endometrial changes, and uterine contractility⁽¹⁶⁾. Unicornuate, arcuate or bicornuate uterus were reported in 0.5-2% of cases⁽¹⁷⁾.

In many cases, the suspicion of congenital uterine malformation can be established during a routine transvaginal ultrasound consultation⁽¹⁸⁾ which can be complemented by 3D ultrasound⁽¹⁹⁾. Hysterosonography or hysterosalpingography, hysteroscopy sometimes, even diagnostic laparoscopy, may be indicated⁽²⁰⁾. The gold standard has remained hysteroscopy combined with laparoscopy for the conclusive diagnosis of uterine malformations⁽²¹⁾. MRI is not routinely used, but is not to be neglected; it was recommended in the evaluation of the urinary tract and possible associated malformations⁽²²⁾.

The assessment and diagnosis of infertility in infertile women should be performed systematically, promptly and cost-effectively, and the causes should be treated as minimally invasively as possible, where feasible⁽²³⁾.

Case report

We reported the case of a 40-year-old woman with a malformed uterus discovered during caesarean section, with a term pregnancy after 15 years of unprotected sexual contact with the same partner. The patient presented to the hospital for painful uterine constrictions that began about 12 hours ago and progressively increased in intensity. We described a compliant patient, who went regularly to the gynecologist for examination, and did all the recommended investigations.

In the 15 years of unprotected sexual intercourse with­out getting pregnant, the patient had ultrasound and blood tests, including AMH (result within normal limits), but she was not evaluated by a doctor specialized in infertility, and she did not perform hysteroscopy, HSG, exploratory laparoscopy or MRI.

During the caesarean section, a detailed evaluation of the pelvic organs and peritoneal cavity was performed after the fetal extraction. No foci of endometriosis, dilatations of the fallopian tubes or fibroid were visualized, but a malformed uterus – bicorn uterus – was noted. Figure 1 depicts an image from the operation when a large uterus was visualized, carrying the pregnancy, and on the left side, the second one, a small uterus.
 

Figure 2 presents the uterine body after the anterior wall has been restored with surjet suture.
 

The operation was successful, both the mother and the newborn had a good evolution. After 72 hours of hospitalization, they left the hospital. The patient left with anticoagulant and analgesic treatment, without any special recommendations compared to other patients operated by caesarean section.

Discussion

Uterine malformations were the consequences of intrauterine abnormalities of combining, cannulation and resorption of the septum during the development of the Müllerian ducts⁽²⁴⁾. Müllerian duct anomalies have been defined as congenital changes diagnosed in many women with recurrent miscarriages. The main manifestations were primary amenorrhea, dysmenorrhea, pelvic pain, endometriosis, sexual difficulties, and low self-regard⁽²⁵⁾. Studies have shown that some of these abnormalities could be treated surgically, others by dilation, and in the case of obstructed menstrual flow, immediate surgical intervention was required⁽²⁶⁾. Surgery for women diagnosed with uterine malformations could significantly improve the reproductive prognosis⁽²⁷⁾.

The present study summarized the accidental discovery of an accessory uterine cavity; this was classified as a Müllerian anomaly. Early diagnosis was found to be crucial in suspicious clinical circumstances, and early treatment significantly improved the patients’ quality of life⁽²⁸⁾.

Endometriosis has been defined as a commonly diagnosed pathology in women of reproductive age which, in addition to pain, has been associated with infertility. The coexistence of endometriosis with uterine congenital anomalies has been frequently associated in the literature, but further research is needed on this topic⁽²⁹⁾.

It has been defined that congenital uterine malformations are the result of defective uterine development. It has been found that the formation of the uterus is a dynamic four-stage process (differentiation, fusion, migration, and canalization), and the resulting uterine malformations are dependent on the stage of uterine formation in which the defect occurs⁽³⁰⁾. Studies in the literature have shown that both channelization and union defects have been associated with low fertility outcomes, low clinical pregnancy rates, and increased rates of early miscarriage⁽³¹⁾.

There are studies available that have examined uterine anomalies in relation to perinatal complications. One study on this topic claimed that the incidence of fetal malpresentation and the frequency of abnormal placental cord insertion were significantly higher in the uterine anomaly group than in the normal uterus group. Examination according to the type of uterine anomaly found large differences in cervical incompetence, malpresentation, caesarean section and abnormal placental cord insertion⁽³²⁾.

Ovarian reserve was strongly associated with women’s fertility potential, which was in direct relation to the quality and quantity of antral follicles and oocytes; certain factors, such as endometriosis, pelvic inflammatory disease, uterine fibroid or the natural aging process, may lead to reduced chances of conceiving. A recent study published in 2023 revealed that the association between uterine abnormalities and uterine fibroid leads to decreased ovarian reserve. The mechanisms involved could be represented by the reduction in the number of antral follicles and the effect on uterine and ovarian blood flow. It has been concluded that the treatment can reduce these effects and improve fertility in affected women⁽³³⁾.

Conclusions

Uterine malformations constitute a significant proportion of the diagnostics in patients of childbearing age, often neglected as a cause of infertility. Early diagnosis and treatment of uterine malformations, when possible, are the main objectives. The risk of miscarriage is not neglected, and the causes are multiple.

Autori pentru corespondenţă: Romina-Marina Sima E-mail: romina.sima@umfcd.ro

CONFLICT OF INTEREST: none declared.

FINANCIAL SUPPORT: none declared.

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