Laparoscopy combined with hysteroscopy in the treatment of Robert’s uterus accompanied by adenomyosis: A case report

Abstract

BACKGROUND

Gynaecologists should be aware of a rare obstructive Mullerian duct abnormality like Robert’s uterus and perform further surgery when necessary.

CASE SUMMARY

We report a 41-year-old mother of two children with Robert’s uterus who was examined and treated by laparoscopy and hysteroscopy. Unlike the existing cases reported in the literature, this patient had a late onset of Robert’s uterus symptoms. Due to right tubal ectopic pregnancy 3 years previously, the patient was treated with right salpingectomy and left tubal ligation but suffered aggravated left lower abdominal pain. She was examined and treated by laparoscopy and hysteroscopy, and is completely asymptomatic at 5-year follow-up.

CONCLUSION

The typical obstructive Mullerian abnormality requires further surgery. Combined laparoscopy and hysteroscopy is an effective, minimally invasive technique with better recovery outcomes than traditional transabdominal procedures.

Key Words: Laparoscopy; Hysteroscopy; Robert’s uterus; Mullerian duct abnormality; Case report

Core Tip: Robert's uterus was found earlier in cases reported in the literature, compared with our case. The blind rudimentary uterine cavity usually communicates with the ipsilateral fallopian tube, permitting partial reflux of retained menstrual blood into the peritoneal cavity. Therefore, the patient’s clinical symptoms were mild. Progressively aggravated dysmenorrhoea occurred due to menstruation retention after sterilisation on the same side of the fallopian tube.

INTRODUCTION

Gynaecologists should be aware of a rare obstructive Mullerian duct abnormality like Robert’s uterus and perform further surgery when necessary. Patients usually present in the postmenarcheal period with unilateral haematometra causing dysmenorrhoea. In our case, no progressive dysmenorrhea after menarche was reported, possibly because the position of the diaphragm was higher and the blind cavity was smaller.

Informed consent was obtained from the patient for publication of this case report and any accompanying images. This report was approved by the Institutional Review Board of our hospital (2021–J-083).

CASE PRESENTATION

Chief complaints

A 41-year-old woman was admitted to the hospital on August 17, 2016, due to left lower abdominal pain for 3 years after sterilisation with aggravation for 1 mo.

History of present illness

The patient reached menarche at the age of 14 years and had regular menstrual cycles with 1-wk duration every 30 d. This patient did not have obvious dysmenorrhoea. Due to right tubal ectopic pregnancy 3 years previously, she had a right salpingectomy and left tubal ligation. However, after the operation, left lower abdominal pain occurred consistently and was aggravated for 1 mo prior to admission.

History of past illness

This patient had two children; one was delivered vaginally, and the other was delivered by caesarean section.

Personal and family history

The patient's personal and family history was unremarkable.

Physical examination

Gynaecological examination revealed the following. The vulva was consistent with a mature, married woman; the vagina was clear; the fornix was empty; the cervix was smooth; the uterine position was forward; the transverse diameter was somewhat wide; the mass was moderate; pain was increasing or changing; activity was normal; the left adnexa area was thickened and tender; and the right adnexa had no obvious abnormality.

Laboratory examinations

The patient's routine blood test and tumour markers carbohydrate antigen (CA)-199 and CA-125 were normal.

Imaging examinations

An ultrasound performed before admission showed a uterine diaphragm (the right corneal angle was connected with the normal uterine cavity, while the left corneal angle was not) (Figure 1A), left fallopian tube effusion (the haematocele was associated with infection), and a left ovarian cystic structure. 3D pelvic ultrasound suggested uterine malformation (Robert’s uterus accompanied by adenomyosis of the left uterine wall) (Figure 1B), left fallopian tube effusion accompanied by left ovary adhesion, and a cystic structure on the left ovary. Urinary tract ultrasound showed that the kidneys, bladder and ureters were normal. Pelvic cavity magnetic resonance imaging (MRI) showed hematometra in a cystic lesion on the left side of the uterus accompanied by adenomyosis of the left uterine wall, and a primarily unicornuate uterus with a noncommunicating rudimentary horn was suspected, which was accompanied by left fallopian tube expansion and a haematocele (Figure 2A and B).

Figure 1 Ultrasound images of Robert’s uterus. A: Two-dimensional; B: Three-dimensional.

Figure 2 Magnetic resonance imaging of Robert’s uterus. A: T2 3D reconstruction image showed endometrioid signals and haematosis between the left muscle wall accompanied by adenomyosis of the left uterine wall T2 3D reconstruction image showed endometrioid signals and haematosis between the left muscle wall accompanied by adenomyosis of the left uterine wall; B: Coronal T2-weighted image showed endometrioid signals and haematosis between the left muscle wall, with a peripheral binding zone, accompanied by adenomyosis of the left uterine wall.

FINAL DIAGNOSIS

According to the patient's symptoms, signs and auxiliary examinations, Robert's uterus, uterine adenomyosis and left fallopian tube effusion were diagnosed.

TREATMENT

The patient underwent laparoscopy and hysteroscopy with tracheal intubation under general anaesthesia on day 5 after admission. On laparoscopy, part of the greater omentum and intestinal canal was densely and vastly adhered to the left adnexa area. The uterus was in a flat position and slightly enlarged. The left side was full and protruded, and the posterior wall was obvious (Figure 3A). The right fallopian tube was absent, but the right ovary was normal in size without an abnormal appearance. The left fallopian tube was distorted and enlarged to 7 cm × 6 cm × 6 cm and was adhered to the left ovary. A cyst of approximately 2.5 cm in diameter with a thin wall and a smooth surface was observed in the left ovary.

Figure 3 Laparoscopy and hysteroscopy. A: Full protrusion at the left side of the uterus observed during laparoscopy; B: An opening was visible only at the right uterine horn and right fallopian tube during hysteroscopy; C: Lesions in the left blind cavity were removed, and the endometrium in the blind cavity was visible during laparoscopy.

Hysteroscopy revealed that the cervix was smooth and the uterus was flat. The uterine cavity was explored to 8 cm deep. Hysteroscopy revealed no abnormalities in the cervix and only the right uterine horn and an opening of the right fallopian tube, but left uterine horn and left fallopian tube opening were not seen. No obvious neoplasm was found in the uterine cavity (Figure 3B). Since the patient did not have a reproductive requirement, we separated the pelvic adhesion by first incising the left fallopian tube and the muscular surface layer at the full blind cavity to the left of the uterus. Adenomyosis lesions were observed. The space between the lesions and normal muscular layer was separated, the adenomyosis lesions containing endometrial tissue were completely removed, Figure 3C, and a 1-0 resorptive suture was used to close the uterus wound.

OUTCOME AND FOLLOW-UP

Postoperative pathology of the uterine tissue noted that smooth muscle tissue and uterine adenomyosis coated the endometrium at the hyperplasia stage. Chronic inflammation was observed in the (left) fallopian tube. At 6-mo and 5-year postoperative follow-up, the patient’s dysmenorrhoea symptoms were resolved.

DISCUSSION

Robert’s uterus is a rare congenital abnormality, and only a few cases have been reported since 1970[1], when it was first described[2,3]. A blind cavity without communication may induce progressive abdominal pain and dysmenorrhoea after menarche due to retention of menstrual material[4]. Robert’s uterus is difficult to diagnose, and its clinical manifestations are similar to those of a uterus with a functional rudimentary horn. Regular inspection methods, such as gynaecological ultrasound, have certain limitations. A type II rudimentary horn with a functional endometrium is difficult to identify as it does not connect with a uterus unicornis; therefore, MRI is the best method to diagnose Robert’s uterus. The contours of the uterus are normal; however, on MRI, two uterine cavities with different sizes can be observed; one of which has a uterine cavity and fallopian tube haematosalpix[5]. Hysteroscopy examination shows an opening on only one side of the uterine horn and fallopian tube. Laparoscopic images show a normal uterus fundus and protrusion on one side, and the fallopian tube on the protruded side is thickened or has a haematocele. If hysteroscopy is combined with laparoscopy, one side of the uterine horn and the opening of the fallopian tube located in the uterine cavity of the nonprotruded side can be detected. The identification points of Robert’s uterus and the rudimentary uterine horn are the bottom of the rudimentary uterine horn and the uterus unicornis, which are separated at dual angles. Our case presented the above laparoscopic and MRI features, and a type II rudimentary uterine horn was identified. Previous reports have shown that the heights of the end points of the inclined diaphragm were different; the higher diaphragm was almost transversal, and the blind cavity was small, containing only a small amount of pink secretion[6], which most likely occurred because the blind rudimentary uterine cavity usually communicates with the ipsilateral fallopian tube, permitting partial reflux of retained menstrual blood into the peritoneal cavity[7]; therefore, the patient’s clinical symptoms are mild. A lower diaphragm can reach the outer opening of the cervix and the blind cavity can be large, resulting in a large amount of blood in haematometra and severe dysmenorrhoea or abdominal pain. In our case, no progressive dysmenorrhoea after menarche was reported, possibly because the position of the diaphragm was higher and the blind cavity was smaller, but progressively aggravated dysmenorrhoea occurred due to menstruation retention after sterilisation on the same side of the fallopian tube, leading to effusion on the same side of the ovary and local adenomyosis lesions on the muscular layer in the blind cavity. Another case reported in the literature included symptoms only several months before admission, and the patient did not have any previous dysmenorrhea or other related uterine haemorrhage. This manifestation may have been caused by a small blind cavity or adhesions from the last caesarean section procedure[8]. Robert’s uterus must be identified by cystic uterine adenomyosis; a key feature is that Robert’s uterus has a normal uterine shape, and only one opening of the uterine horn and fallopian tube is evident, as in our case.

The traditional surgical treatment for Robert’s uterus is incision of the diaphragm through an abdominal approach between the uterus and focal blind cavity with a normal uterine cavity. Then, the uterus is sutured to restore the normal anatomy as closely as possible. Studies have shown that hysteroscopy combined with laparoscopy may be helpful for the diagnosis and classification of female genital system anomalies and surgical procedures[9,10]. Some researchers[2,3] have also suggested that the use of hysteroscopy and laparoscopic surgery for Robert’s uterine septum resection presents some difficulties, and hysteroscopy and laparoscopy combined with ultrasound may be a better method to diagnose and treat Robert’s uterus. These methods, especially in patients with fertility requirements, can substantially reduce surgical trauma and facilitate a shorter operation time, reduced bleeding and faster postoperative recovery and can protect the postoperative integrity of the uterus[2]. In our case, no progressive dysmenorrhoea after menarche was reported, possibly because the position of the diaphragm was higher and the blind cavity was smaller. The patient had uterine adenomyosis appeared on the muscular layer of the blind cavity because of blood retention during menstruation after ipsilateral tubal sterilisation. She had no reproductive requirement. Therefore, the patient consented to removal of uterine adenomyosis and the endometrium of the blind cavity as well as an ipsilateral salpingectomy by hysteroscopy and laparoscopy after a clear diagnosis was established.

CONCLUSION

There is no need to correct the uterine deformity surgery for the patient without fertility requirement, only the symptoms of menstrual blood obstruction. Hysteroscopy and laparoscopy are the suitable methods for diagnosis and treatment. According to the different conditions and the requirements of patients, individualised surgical treatments were selected. The combination of laparoscopy and hysteroscopy is an effective, minimally invasive technique with better recovery outcomes than traditional transabdominal procedures.