Chronic Non Pueperal Inversion Of Uterus Secondary To Submucosal Fundal Myoma

Author Information

Bakre Tejashree*, Gupta AS**, Hira Priya***, Parulekar SV.****

(* Third Year Resident, ** Professor, **** Professor and Head, Department of Obstetrics and Gynecology; *** Additional Professor, Department of Radiology, Seth G.S. Medical College & K.E.M. Hospital, Mumbai, India)

Abstract

A case of  chronic inversion of uterus secondary to a large submucous fundal fibroid in a 40 year old multiparous woman with previous 2 lower segment cesarean sections (LSCS) is presented. Kustners' procedure was used to correct the inversion. Correction was preceded by vaginal myomectomy and followed by hysterectomy.

Introduction

Non puerperal uterine inversion is a very rare clinical entity. Almost all non puerperal cases are chronic and only around 10% have acute presentation.^[1] Chronic non puerperal inversion of uterus is associated with uterine tumors in about 90% of cases, most common being submucous fibroid.^[2]

Case Report

A 40 year old woman, para 2 living 2 with history of previous 2 lower segment cesarean sections, presented with complaints of something coming out of the vagina accompanied with bleeding since 15 days and lower abdominal heaviness. There were no voiding difficulties. She had a history of menometrorrhagia for past 2 months. There were no other surgical and medical high risk factors. On examination she had severe pallor and tachycardia. Abdomen was soft. There was a Pfannenstiel scar. On local examination a large, elongated, congested, red, soft mass 15 x 4 x 4 cm in size, with a firm, well demarcated, white lesion about 6 x 5 cm in dimension was seen at the end of the elongated mass. It was devoid of any punctum and was lying outside the introitus. The mass could be incompletely reduced  into the vagina.

Figure 1. The Prolapsed mass with the fibroid, as seen in the outpatient clinic

Figure 2. The Prolapsed mass with the fibroid, as seen in the operation theater. The leiomyoma is pointed out by blue arrows and the cervical ring by solid black arrows.

On vaginal examination a cervical rim without any depth could be felt all around except posteriorly. A provisional diagnosis of uterine inversion with a submucous fibroid polyp was made. The prolapsed part  was reposited back in the vagina and restrained by a vaginal tampon soaked in glycerin acriflavin solution.  However, it could not be kept reduced in the vagina.  The patient developed  fever 48 hours after admission. Pus was seen oozing from the inverted endometrium or the presumed stalk of the fibroid. A wound swab was sent for culture and sensitivity. It grew Pseudomonas areuginosa sensitive to Levofloxacin. The patient was given oral Levofloxacin for 10 days. Daily dressings with povidone iodine were performed.  The fever settled within 72 hours of  starting Levofloxacin. Her serological and biochemical preoperative investigations, chest radiography and electrocardiogram were done.  Hemoglobin was 6 g/dL.  All other investigations were in their normal range. The patient was transfused with 3 units of packed cells over 3 days. Ultrasonography revealed indentation of fundus with fundal fibroid (4.2 x 3.5 cm). Computerized tomography (CT) scan showed  cupping of fundus with inversion of uterus and a submucous fundal fibroid 5.5 cm in diameter. Magnetic resonance imaging (MRI) confirmed the findings showing U shaped uterine cavity on saggital images with complete inversion , a submucous fundal fibroid 5.5 cm in diameter with ovaries present in midline at site of inversion but without bladder involvement.

Figure 3. CT Scan showing the inverted fundus, ovaries in the cup and the cervical ring.

Figure 4. MRI Scan confirming CT scan findings.

The patient was operated under spinal anesthesia.  The cervical ring was very vascular anteriorly. Spinelli's surgery was not considered due to this vascularity, chances of presence of bladder in the inverted mass and adhesions between the uterus and bladder due to the previous surgeries. Kustner’s procedure was performed. The cervical ring was divided vertically in midline posteriorly. Divided edges of the cervix were held with Allis’ forceps. The myoma was enucleated from its bed in the fundus because the inverted uterus could not be reposited with the myoma in situ. The incision was extended in the isthmus and the uterine body posteriorly to facilitate correction of the uterine inversion.  The correction of the inversion was confirmed by the visualization of the cervix in central position and the anterior and posterior  fornices in front of and behind it respectively. Vaginal hysterectomy was then done by the standard method. Postoperative period was uneventful and patient was discharged on day 7. The perioperative and the post operative period was covered by antibiotics.

Figure 5. Kustner's operation: the posterior ring of the cervix is defined (arrow).

Figure 6. Kustner's operation:. Incision of posterior cervical ring and identification of the edges of the cervix (arrows).

Figure 7. Kustner's operation: myomectomy.

Figure 8. Kustner's operation: excised myoma (M) and its bed (B).

Figure 9. Kustner's operation: reformation of the lips of the cervix. A: anterior lip of cervix, P: two ends of the divided posterior lip of cervix.

Discussion

Uterine inversion is very uncommon.^[3,4] Most of the cases occur in the puerperal period. Non puerperal cases are mostly chronic and account for only 15% of all cases of inversion.^[1]  Over 90% of these are associated with uterine tumors, of which 20% are malignant.^[1]

Chronic inversion is of two types, incomplete and complete. Incomplete is the type where fundus protrudes through the cervix but lies inside the vagina and complete is one in which whole of the uterus including the cervix are inverted and lie outside the introitus. Vagina may also be involved in latter type.^[5] Predisposing factors for uterine inversion include submucous fibroid, endometrial polyp and uterine sarcoma.^[2] Most cases occur secondary to  prolapsed fibroid, with some reports stating that the fibroid was infected and necrotic.^[4]  The most common presenting symptoms of chronic inversion are chronic vaginal discharge and irregular vaginal bleeding leading to anemia.^[5] In this case the patient presented with a mass coming out per vaginum with foul smelling discharge from the mass and fever secondary to necrosis and inflammation of fibroid and surrounding endometrium. As the entity is rare a high index of suspicion is required for diagnosing uterine inversion. It can be mistaken for other conditions like prolapsed uterine fibroid, uterine sarcoma, uterovaginal prolapse and endometrial polyp.^[3,4,5]   Many a times the diagnosis is only made intraoperatively.^[6]  This leads to surgical difficulties, increase in complication rates and the surgeon may not be able to choose the most appropriate surgical method. The recto abdominal examination is often the most diagnostic clinical method wherein on bimanual palpation dimpling of uterine fundus is felt. Clinical diagnosis is often aided by imaging modality. USG is often the first modality for diagnosing an inversion of the uterus. Indentation of the fundal area, a depressed longitudinal grove extending from the uterus to the center of the inverted fundus and target sign with hyperechoic fundus surrounded by a hypoechoic rim, suggesting fluid occupying the space between the inverted uterine fundus and the vaginal wall are signs described in relation to the chronic uterine inversion.^.[5]  MRI and CT scan are useful diagnostic tools. They also help in understanding the extent of involvement of the bladder and ovaries in the inverted tissues.^[1] Signs indicative of uterine inversion on MRI are U shaped uterine cavity and a thickened and inverted uterine fundus on a sagittal image and a “bulls-eye” configuration on an axial image.^[1,3,4] Surgical treatment of chronic uterine inversion depends upon patient's fertility, stage of inversion and associated pathology.^[3] There are many abdominal and vaginal surgical approaches  to correct inversion.^[4] The vaginal and abdominal approaches have their own advantages and disadvantages. Hence opinion is divided regarding the management of chronic inversion of the uterus. As a leiomyoma was  present and uterine inversion was complete the vaginal approach was preferred in the case described. Vaginal myomectomy was done to debulk the uterine mass prior to  the correction of the inversion. The divided cervix is sutured after the correction of the inversion if the uterus is to be conserved. If a hysterectomy is to be performed, reposition is always done first, because hysterectomy on the inverted uterus is associated with difficulty in identification of the peritoneal pouches, and also associated with a great risk of injury to the urinary bladder and ureters.^[2]

Spinelli’s and Kustner ‘s operations are similar vaginal procedures, the only difference being that Spinelli's approach is anterior and involves division of the cervical ring, while Kustner's is a posterior approach with incision on the posterior cervical ring.^[2,3] Kustner’s approach was preferred here as there is little risk of injuring the bowel, because of the deep reflection of the pouch of Douglas and anterior approach involved danger of damaging the bladder during separation, risk being more due to complete inversion.^[7] The risk was even more in our case due to fibrosis and loss of tissue planes due to previous cesarean scars. Surgical reposition can also be done abdominally by Huntington’s procedure wherein round ligaments are pulled on and the inverted part of the uterus is pulled back into the peritoneal cavity by pulling on a series of Allis’ forceps applied progressively at lower levels. In Haultain’s  procedure, done transabdominally the constricting ring is divided posteriorly by  a vertical incision and then the inversion is corrected.^[3] In Dobbin’s procedure, the cervical ring is divided in midline anteriorly by an abdominal approach and then the inversion is corrected.^[3]

Conclusion

These rare cases pose diagnostic and management challenges. A high index of suspicion by the clinician and the radiologist can detect the true nature of the condition.  Once a diagnosis is established the surgical correction can be done by  adopting the best option from the various techniques described in literature. As the clinical entity of chronic inversion is rare definite management guidelines cannot  be recommended and management of each case has to be individualized.

References

1. Ashraf-Ganjooie T: Nonpuerperal uterine inversion: A case report. Archives of Iranian Medicine 2005;8(1):63-66  .
2. Kagne SS, Thawal YA, Tambe SG. An extremely rare case of chronic non-puerperal uterine inversion treated by myomectomy preceding vaginal hysterectomy. Journal of evolution of medical and dental sciences. 2013;46: 8976-8979.
3. Shabbir S, Ghayasuddin M, Younus SM, Baloch K. Chronic non puerperal uterine inversion secondary to sub-mucosal fibroid: JPMA 2014;64:586-588.
4. Kilpatrick CC, Chohan L and Maier RC. Chronic nonpuerperal uterine inversion and necrosis: a case report. J Med Case Reports 2010;4: 381.
5. Jain S, Aherwar R, Joshi P. Chronic Non-Puerperal Uterine Inversion; Fibromyoma Uteri as a Cause- A Case Report.  Sch J Med Case Rep 2014;2(2):100-102.
6. Sharma JB, Kumar S, Rahman SM, Roy KK, Malhotra N. Non-puerperal incomplete uterine inversion due to large sub-mucous fundal fibroid found at hysterectomy: a report of two cases. Archives of Gynecology and Obstetrics 2009;279(4):565-567.
7. Culiner A, Charlewood GP. The surgical management of chronic inversion of the uterus. S. A. Medical Journal May 1954; 459-60.
8. Crossen HS.Chronic inversion of the uterus. In Operative Gynecology. 1st ed.  St. Louis: CV Mosby Co; 1917: Pp. 151-160.

Citation

Bakre T, Gupta AS, Hira P, Parulekar SV. Chronic Non Pueperal Inversion Of Uterus Secondary To Submucosal Fundal Myoma. JPGO 2014. Volume 1 Number 10. Available from: http://www.jpgo.org/2014/10/chronic-non-pueperal-inversion-of.html