Traumatic transection of the uterus: missed, diagnosed, and repaired-a case report and review of literature.

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Abstract

BackgroundUterine transection is a very rare result of pelvic trauma, with less than a dozen case reports in the literature. The reporting radiologist's unfamiliarity with this injury led to a missed diagnosis on ultrasound, as well as on magnetic resonance imaging. This led to delayed treatment. We hope that with this publication, a wider audience will consider this possibility in the appropriate clinical and imaging setting.Case presentationWe report the case of a 25-year-old South Asian woman with primary amenorrhea, mild irregular spotting, severe monthly pain, and primary infertility, who had uterine transection. She sustained trauma at the age of 2 years. She was worked-up for infertility, including ultrasound and magnetic resonance imaging; however, the radiologist's reports were erroneous. On the final ultrasound, and later on magnetic resonance imaging, the cervix with an open internal os was seen lying separate from the corpus. Hematometra and hemoperitoneum were also noted, and a diagnosis of transection of the uterus was made. At surgery, a uterine transection, as suggested by imaging, was found. The two separated components of the uterus were aligned and anastomosed. The patient, since her repair has had two menstruation cycles, on time and pain free.ConclusionThe rarity of the condition and unfamiliarity with the pathology caused the initial misdiagnosis. If the injury remains undiagnosed it causes infertility, severe amenorrhea and/or dysmenorrhea, and a markedly impaired quality of life. The injury once diagnosed is easily repaired and often results in the return of menstruation and, sometimes, even fertility.
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Patient

A 25-year-old married, Pakistani woman was being worked-up for primary infertility and primary amenorrhea. She had a history of monthly pelvic pain and irregular spotting but never experienced regular menstruation. History was significant for blunt abdominal trauma at the age of 2 years, when a low-height, under-construction wall collapsed upon her; she had lost consciousness for a few minutes but never needed any surgical treatment. She had well-developed secondary sexual characteristics but had had no pregnancy after 5 years of marriage. She had been investigated for primary infertility, and her hormone profile of follicle stimulating hormone (FSH), luteinizing hormone (LH), thyroid stimulating hormone (TSH), and prolactin was normal; her anti-mullerian hormone (AMH) level was also normal. Previous ultrasound and magnetic resonance imaging (MRI) reported a hypoplastic uterus with a blind horn. Both were done by the same radiologist. She was eventually referred to our center where an ultrasound was repeated. Both transabdominal as well as transvaginal ultrasound were performed. Ultrasound showed the corpus of the uterus to be separate from the cervix. The corpus was exceptionally mobile, and on transvaginal ultrasound, it could be easily pushed away by the ultrasound probe. The cervix was intact, but the internal os of the cervix was open (Fig.  1 ); there was a moderate amount of complex fluid distending the uterine canal of the corpus. A moderate amount of complex fluid was observed in the pelvis. Fig. 1 Transvaginal ultrasound showing the corpus to be completely separate from the cervix (Cx). The corpus was filled with complex echogenic fluid, hematometra (Hm). The cervix (Cx) was in its normal location, the internal os, marked by the curved red arrow, was open. A moderate amount of fluid, hemoperitoneum (Hp), was seen in the pelvis Transvaginal ultrasound showing the corpus to be completely separate from the cervix (Cx). The corpus was filled with complex echogenic fluid, hematometra (Hm). The cervix (Cx) was in its normal location, the internal os, marked by the curved red arrow, was open. A moderate amount of fluid, hemoperitoneum (Hp), was seen in the pelvis There was a 6-cm left ovarian cyst. A left-sided dilated tubular structure was also noted. A uterine transection (cervico-isthmic disjunction) with hematometra of the corpus, hemoperitoneum, and an open internal os communicating with the peritoneum was diagnosed. An MRI was subsequently done (Fig.  2 ), which showed the same findings; the internal os was clearly seen to be open. Fig. 2 Magnetic resonance image: there is separation of the corpus (Utc) from the cervix (Cx), the internal os (curved arrow) is open and communicates with the hemoperitoneum (Hp). The corpus is distended with blood (Hm) Magnetic resonance image: there is separation of the corpus (Utc) from the cervix (Cx), the internal os (curved arrow) is open and communicates with the hemoperitoneum (Hp). The corpus is distended with blood (Hm) At surgery, the cervix was identified by passing a sound, and then a catheter on a guide wire through the external os. It passed without any resistance into the peritoneum, confirming that the internal os was open. The corpus was supplied by the ovarian artery in the infundibulopelvic ligament. The corpus was drained and the caudal end was identified and freshened; similarly, the upper end of the cervix was freshened. The catheter, already in the cervical canal, was pushed into the corpus (Fig.  3 ), and the two were attached with interrupted sutures in two layers. Fig. 3 Surgical field. The corpus (Utc) is separate from the cervix (Cx), a catheter (Cath) has been passed from the cervix to the corpus Surgical field. The corpus (Utc) is separate from the cervix (Cx), a catheter (Cath) has been passed from the cervix to the corpus A moderate amount of hemoperitoneum was also drained. The catheter within the uterine canal was removed after 3 weeks. The patient was examined by ultrasound 1 week later (Fig.  4 ). The cervical canal and uterine canal were aligned; there was a moderate amount of complex fluid in the uterine canal. A small-to-moderate amount of free fluid was also noted in the peritoneum. The patient has had two normal and painless periods since the surgery; the first 1 week after the catheter removal, and the second a month after the first period. Fig. 4 Ultrasound 1 month after surgery. The cervical and uterine canals are aligned (arrows), there is a moderate amount of complex collection in the uterine canal, which has both anechoic (*) as well as echogenic (**) components. The circumferential stitches used for repair are visible (open arrows) Ultrasound 1 month after surgery. The cervical and uterine canals are aligned (arrows), there is a moderate amount of complex collection in the uterine canal, which has both anechoic (*) as well as echogenic (**) components. The circumferential stitches used for repair are visible (open arrows) This was the first time in her life that menstruation was not accompanied by excruciating pain.

Conclusion

We believe that this type of injury might be more common than reported and the paucity of reports might, in part, be due to misdiagnosis as well as the rarity of the injury. The purpose of this paper is to highlight the existence of this rare injury to a larger audience and report the possibility of repair in a low-resource setting.

Discussion

Uterine transection is a very rare injury with only a few case reports in the literature. It has been variably called cervico-isthmic disjunction, cervico-isthmic separation [ 1 , 2 ], or uterine avulsion [ 3 , 4 ]. Uterine avulsion has also been used to describe the complete or nearly complete separation of the whole uterus from its attachments [ 5 ]. Most cases are associated with high-intensity trauma involving fracture of the pelvis and injury to the bladder [ 1 , 6 ]; many cases involve a gravid uterus [ 5 , 7 ]. Motorcar accidents [ 8 ] and seatbelt injuries have been implicated in this type of rupture [ 7 ]. Iatrogenic injury has also been associated with cervico-isthmic separation and has been reported as a complication of myomectomy, adenomyomectomy, and ovarian surgery for teratomas [ 9 – 11 ]. In one case report, cervico-isthmic disjunction was thought to be congenital [ 12 ] but the paper did not mention the absence of trauma. There are very few cases, like ours, that involve trauma in infancy or early childhood that did not need any special surgical management [ 2 ]. The initial response to such injury is often hysterectomy but repair of traumatic, noniatrogenic transection has also been reported, though very rarely, and only seven cases were found up until 2023 [ 2 ]. Our case might be the eighth to be reported. In our case, the corpus was entirely separate from the cervix, and the internal os was open and communicated with the peritoneum. There was a moderately large hemoperitoneum that might have been the result of retrograde menstruation, as has already been reported [ 13 ]. Endometriosis, presumably by the same mechanism, is present in most of the cases of uterine transection, especially in old cases [ 2 ]. Despite the dramatic appearance of the injury, repair is not technically difficult and should be the preferred option instead of hysterectomy. Adequate suturing in layers after alignment, usually with a catheter or dilator extending from the cervix to the corpus, restores menstrual function as well as fertility in many of these cases [ 1 , 2 ]. Both immediate and delayed repair have good results. Laparoscopic repair has the advantages of a magnified surgical field. Our patient has had two, normal, relatively painless menstrual cycles about a month after surgery and then a month after that. This was the first time that menstruation was not associated with excruciating pain. The presence of uterine fluid noted on the ultrasound, which was performed a week after the first periods, probably represents evolving stenosis at the repair site. This is a known and frequent complication [ 2 ]. The patient presented with infertility and cyclical pain and was worked-up with ultrasound and MRI. Initial ultrasound and MRI examinations erroneously reported unicornuate uterus with a blind horn. We believe the ultrasound and MRI were extremely suggestive of the correct diagnosis, but the reporting physician was unaware that this injury exists and was a victim of “anchoring or framing bias,” wherein an impression made early in the diagnostic process becomes a firm, unchanging opinion, and alternate possibilities are not considered [ 14 ]. This type of erroneous reporting, however, is not unique to our case, and in many of the publications, the diagnosis became obvious only during direct visualization, either at laparoscopy or laparotomy [ 8 , 15 ].

Introduction

Uterine transection is often the result of extensive high-intensity trauma. Only a few case reports exist in the English language literature. The rarity of the condition makes diagnosis difficult if the imaging physician is unaware of this injury. Many times, the transection is diagnosed upon surgery. Since most cases involve young women, it is desirable to attempt a repair rather than take the easier path of hysterectomy. A prior diagnosis is important to plan the surgery at a center where this repair can be undertaken by an experienced surgeon.

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