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
- Ashraf-Ganjooie T: Nonpuerperal uterine inversion: A case report. Archives of Iranian Medicine 2005;8(1):63-66 .
- 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.
- Shabbir S, Ghayasuddin M, Younus SM, Baloch K. Chronic non puerperal uterine inversion secondary to sub-mucosal fibroid: JPMA 2014;64:586-588.
- Kilpatrick CC, Chohan L and Maier RC. Chronic nonpuerperal uterine inversion and necrosis: a case report. J Med Case Reports 2010;4: 381.
- 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.
- 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.
- Culiner A, Charlewood GP. The surgical management of chronic inversion of the uterus. S. A. Medical Journal May 1954; 459-60.
- 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