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ABSTRACT An isthmocele appears as a fluid pouchlike defect in the anterior uterine wall at the site of a prior cesarean section and ranges
in prevalence from 19% to 84%, a direct relation to the increase in cesarean sections performed worldwide. Many defini-
tions have been suggested for the dehiscence resulting from cesarean sections, and we propose standardization with a single
term for all cases—isthmocele. Patients are not always symptomatic, but symptoms typically include intermittent abnormal
bleeding, pain, and infertility. Pregnancy complications that result from an isthmocele include ectopic pregnancy, low im-
plantation, and uterine rupture. Magnetic resonance imaging and transvaginal ultrasound are the gold standard imaging techniques
for diagnosis. Surgical treatment of an isthmocele is still a controversial issue but should be offered to symptomatic women
or the asymptomatic patient who desires future pregnancy. When surgery is the treatment choice, laparoscopy guided by
hysteroscopy, hysteroscopy alone, or vaginal repair are the best options depending on the isthmocele’s characteristics and
surgeon expertise. Journal of Minimally Invasive Gynecology (2018) 25, 38–46 © 2017 AAGL. All rights reserved.
Keywords: Cesarean scar dehiscence; Hysteroscopy; Laparoscopy; Vaginal repair
It has been 30 years since the World Health Organiza- first laparoscopically by Jacobson et al in 2003 [7]. The first
tion released a statement warning about the high rate of scar defect ever reported was in 1975 when Stewart et al [8]
cesarean sections [1] and recommending a maximum 15% noted that preoperative hysterography or pelvic arteriogra-
rate of surgical intervention. Despite this, the United States phy might help with diagnosis and that the uterus could be
reported an increase of 50% in cesarean sections from 1996 saved by excision of the lower segment scar. The world-
to 2007 [2], and other countries such as Brazil reported an wide prevalence of isthmoceles out of all cesarean sections
overall cesarean section rate of 45% and a private practice ranges from 19% to 84% [9–13], but this may be underes-
rate of 81% [3]. The choice of delivery method is a complex timated because of asymptomatic patients and a lack of
topic based on physical and psychological health, social and clinician awareness. Sonohysterography (SHG) identified a
cultural context, and quality of maternity care. However, it higher number of patients with isthmoceles (56%–78%) than
is imperative for patients to be informed of the risks of both transvaginal ultrasound (24%–69%) as noted by van der Voet
vaginal and cesarean deliveries [4]. et al [14].
An isthmocele, also called a niche, cesarean scar defect,
or cesarean scar dehiscence, is a pouchlike defect of the an-
terior uterine isthmus at the site of a prior cesarean section Symptoms
[5], which was first described by Morris in 1995 [6] and treated Many patients with isthmoceles are asymptomatic, and pa-
tients might consult with different physicians before the correct
The authors declare that they have no conflict of interest. diagnosis is found. The most frequent complaint relates to
Corresponding author: João Alves, MD, Hospital da Luz, Avenida Lusíada, intermittent postmenstrual bleeding. The isthmocele func-
100, Lisboa, 1500-650 Lisboa, Portugal. tions as a reservoir collecting blood during menstruation, with
E-mail: jmiguelalves@gmail.com irregular menses that can persist from 2 to 12 days [15].
Submitted June 15, 2017. Accepted for publication September 5, 2017. Vervoort et al [16] and van der Voet et al [14] reported that
Available at www.sciencedirect.com and www.jmig.org 28.9% to 30% of patients with isthmoceles experienced
1553-4650/$ — see front matter © 2017 AAGL. All rights reserved.
https://doi.org/10.1016/j.jmig.2017.09.022
Setubal et al. A Review of Uterine Isthmocele Treatment 39
abnormal bleeding compared with 6.9% of patients without English, French, Spanish, and Portuguese languages were se-
it. In addition, women with an isthmocele defect greater in lected. All published articles with a clinical description of the
size than half of the adjacent myometrium reported abnor- treatment of an isthmocele were selected, excluding case
mal uterine bleeding more frequently [17]. Because of other reports, review articles, and video reports. Video and case
more common causes of irregular menstruation, clinicians reports of treatment not previously described were in-
might not identify this specific symptom to be related to the cluded. Two-hundred fifteen studies were found. We excluded
scar defect. 175 studies that did not address isthmocele management; we
Several authors [10,18,19] have described isthmoceles as excluded another 10 studies because they were review ar-
a cause of infertility, stating deficient sperm motility and im- ticles or previously published video or case reports. Two case
plantation. Pain and dysmenorrhea are general symptoms reports were included because of their innovative descrip-
common to numerous gynecologic causes. The relationship tion of isthmocele management (Fig. 2). The studies were
between isthmoceles and pain is not clear but could be related analyzed according to the Preferred Reporting Items for Sys-
to abnormal myocontraction caused by physiological irregu- tematic Reviews and Meta-Analyses statement and The
larities and continuous efforts of the uterus to empty the Cochrane Collaboration’s Risk of Bias Tools for Non-
contents of the isthmocele. Wang et al [20] found a signifi- Randomized Studies.
cant relationship among dysmenorrhea, the width of the defect,
and abnormal bleeding. Van der Voet et al [14] reported a
97% improvement in pain when isthmoceles were resected Results
hysteroscopically. The incidence of ectopic pregnancies im- Tables 1 and 2 describe all included studies and the type
planted in the myometrium at the site of the isthmocele is of treatment for each study [5,16,18,24–50].
reported to be approximately 1 in 1886 to 2216 pregnancies
[21–23]. With the development of the fetus and expansion
of the gestational sac, the walls of the isthmocele might Isthmocele Treatments
rupture, presenting a risk to both the mother and the fetus.
There are more than 30 options to treat ectopic pregnancy Medical Treatment
in the myometrium including methotrexate injection; removal Zhang et al [26] have published a study on the use of oral
by means of the vaginal, hysteroscopy, laparotomy, or lapa- contraceptives with estrogen and progesterone in 18 pa-
roscopy approach; or hysterectomy [23]. The choice depends tients and have found it to be effective regarding the duration
on the patient’s desire for future fertility and team expertise. of menses (reducing from 10 days to 5 days). Other authors
[24,25] have also described oral contraceptives to be effec-
tive in reducing bleeding disorders. Florio et al [25] have
Imaging described less bleeding and less pain after the use of oral con-
An isthmocele is typically diagnosed on transvaginal traceptives or hysteroscopic resection with better results in
sonography or SHG and appears as a wedge-shaped an- women undergoing hysteroscopic correction compared with
echoic area with a depth of at least 1 mm and an indentation oral contraceptives. The use of an intrauterine device with
of the myometrium of at least 2 mm in the uterine isthmus levonorgestrel (Mirena; Bayer, Whippany, NJ) has not shown
at the cesarean section scar site [19]. Ultrasound is used to a benefit in these women [26], but a short follow-up of only
measure the depth and size (longitudinal) of the dehiscent 6 months was reported. The main disadvantage to using oral
scar and the thickness of the residual myometrium covering contraception is avoiding pregnancy. Thus, the best medical
the dehiscence. The prevalence of cesarean scar defects in treatment for women with isthmoceles who do not want to
randomly selected women with previous cesarean sections become pregnant seems to be oral contraception.
was found more frequently with SHG compared with trans-
vaginal sonography (56%–84% vs 24%–70%) [19]. Vaginal
Magnetic resonance imaging is useful to evaluate the thick- Surgery by means of the vagina has been described by
ness of the lower uterine segment, the depth of the isthmocele, Zhang et al [26,42] and others [33,37,39,46–48]. According
and the content of the endometrial and niche cavity (Fig. 1). to Zhang et al [26,42], once the cervix was grasped by 2
It allows the evaluation of other associated pathologies such forceps, diluted bovine pituitary hormones were injected
as adenomyosis or adnexal, uterine, or pelvic diseases. subepithelially at the cervicovaginal junction to determine
tissue planes and reduce minor bleeding. The incision was
made where the hormone was injected, and the bladder was
Methods
then deflected off the anterior cervix. Once identified at the
We searched PubMed, Cochrane Review, Clinical Trials, uterine isthmus, the isthmocele was excised and repaired.
and Scholar Google for studies published between 1980 and Zhang [42] has reported no complications (14 patients). This
July 2017 using the following keywords and Medical Subject technique requires surgical expertise to avoid damaging the
Headings: isthmocele, cesarean scar defect, uterine scar defect, surrounding organs. It also necessitates that the isthmocele
pouch, sacculation niche, and surgery. Articles published in is not too high or vaginal correction would be difficult. Results
40 Journal of Minimally Invasive Gynecology, Vol 25, No 1, January 2018
Fig. 1
A magnetic resonance image of an isthmocele. On the left is the sagittal T2 view of the female pelvis with the thickness of the anterior uterine wall. On
the right are axial and sagittal T2 sequences with longitudinal, anteroposterior, and transversal diameters of the saccular defect.
show that menstruation duration diminishes after treatment hemostasis [49]. Other groups have reported the same pro-
[26] and myometrial thickness increases [42]. cedure with good clinical results (i.e., a shorter duration of
Most patients have symptom relief (reported between 85% menstrual period and improved fertility) [39,50]. No com-
and 93%) and minor if any complications after this surgical plications have been noted [39,49], and Jeremy et al [39] have
approach. Xie et al [33] and Zhang et al [26,42] have noted described a 71% pregnancy rate after laparotomy.
a resulting 22% pregnancy rate.
Hysteroscopy
Laparotomy Several articles have reported hysteroscopic treatment of
Schepker et al [49] have described laparotomy as a com- isthmoceles [5,16,18,25–35]. Xie et al [33] have reported 1
plete resection of the dehiscent myometrium and a 2-layered of the largest series with 77 patients with hysteroscopic treat-
suture with 2-0 to 4-0 Vicryl interrupted sutures (Ethicon, ment. Using monopolar electrical current for cutting and
Somerville, NJ). It is important to correct the myometrial coagulation and sterile preparation, the cervix was dilated to
wound margins layer by layer (“‘edge-to-edge’”) as well as 12 mm. The hysteroscope (Figs. 3 and 4) was then introduced
Setubal et al. A Review of Uterine Isthmocele Treatment 41
Fig. 2 Table 1
The Preferred Reporting Items for Systematic Reviews and Meta-
The Type of Treatment for Isthmoceles
Analyses flowchart.
Type of Treatment Studies
Records identified through database search
n = 215
Expectant Vervoort et al, 2015 [16]
Articles excluded Hormonal Tahara et al, 2006 [24]
n = 175
Florio et al, 2011 [25]
Zhang et al, 2016 [26]
Full text articles assessed for eligibility Hysteroscopic Gubbini et al, 2008 [5]
n = 40
Vervoort et al, 2015 [16]
Excluded review Gubbini et al, 2011 [18]
articles, case reports,
and video reports Fabres et al, 2005 [27]
n = 10 Chang et al, 2009 [28]
Studies included in the review Florio et al, 2011 [25]
n = 30
Wang et al, 2011 [29]
Feng et al, 2012 [30]
Li et al, 2014 [31]
Perez-Medina et al, 2014 [32]
under direct visualization, and after determining the loca- Xie et al, 2014 [33]
tion of the isthmocele, a cutting loop was used to remove the Raimondo et al, 2015 [34]
fibrotic tissue flap under the pouchlike defect, from the bottom Tanimura et al, 2015 [35]
of the defect to the endocervical canal. The remainder of the Zhang et al, 2016 [26]
pouch was cauterized [33]. Hysteroscopic treatment seems Laparoscopy Donnez et al, 2008 [36]
Klemm et al, 2005 [37]
useful in cases of infertility, as described by Tanimura et al
Yalcinkaya et al, 2011 [38]
[35]. No complications have been noted although Raimondo
Jeremy et al, 2013 [39]
et al [34] have reported 16 of 120 patients with the same symp- Marotta et al, 2013 [40]
toms postoperatively. Li et al, 2014 [31]
Tanimura et al, 2015 [35]
Laparoscopy Nirgianakis et al, 2016 [41]
The first laparoscopic repair of a uteroperitoneal fistula Zhang et al, 2016 [26]
Zhang, 2016 [42]
caused by cesarean section was performed by Jacobson et al
Liu et al, 2016 [43]
[7]. A skilled laparoscopic surgeon can use conventional lapa-
Akdemir et al, 2017 [44]
roscopy or robotic-assisted surgery to correct an isthmocele. Donnez et al, 2017 [45]
The surgical technique (Figs. 5–7) was described by Donnez Vaginal Klemm et al, 2005 [37]
et al in 2008 [36], and his group has recently published a large Luo et al, 2012 [46]
prospective series with 38 patients [45]. The surgical tech- Jeremy et al, 2013 [39]
nique was described as using carbon dioxide laser, the scar Chen et al, 2014 [47]
was opened from one end to the other, and fibrotic tissue was Xie et al, 2014 [33]; Zhang et al, 2016 [26]
excised from the edges of the defect to access the healthy Zhang, 2016 [42]
myometrium [45]. Before closing, a Hegar probe (Skalar Sur- Zhou et al, 2016 [48]
gical Instruments, West Chester, PA) was inserted into the Levonorgestrel system Zhang et al, 2016 [26]
Laparotomy Jeremy et al, 2013 [39]
cervix to preserve continuity of the cervical canal with the
Schepker et al, 2015 [49]
uterus [45]. Multiple layers of separate sutures were used to
Pomorski et al, 2017 [50]
achieve double-layer closure, and the peritoneum was then
closed [45].
The critical step of this procedure is to correctly identify
the isthmocele, which can be accomplished using the fol- endocervical canal and forwarded blindly at the level of the
lowing techniques: hysteroscopy to evaluate the uterine cavity uterine isthmus. This maneuver distended the isthmocele on
and the defect; if the isthmocele is not identified by laparos- the uterine wall. The continuing pressure led to a “hooking
copy, hysteroscopy can be repeated; and the tip light of the effect,” which defined the defect more, allowing it to be per-
hysteroscopic instrument can be inserted into the defect. forated under laparoscopic visualization. This method has been
Donnez et al [45] used the dimmed light of the laparo- described by Api et al [51] in a case report and named the
scopic instrument concomitantly to illuminate the “slip and hook technique” to locate the isthmocele. After-
hysteroscopic light through the scar. If at this point the ward, the scar tissue can be completely excised and the dilator
isthmocele was not correctly identified, a number 6 Hegar pulled back gently as a guide for suturing the myometrium
dilator (Skalar Surgical instruments) was placed in the and preserving the continuity of the cervical canal (Figs. 5–7).
42
Table 2
A Description of the Articles Found
43
44 Journal of Minimally Invasive Gynecology, Vol 25, No 1, January 2018
Fig. 3 Fig. 5
The location of the isthmocele by hysteroscopy. With bipolar resectoscopy, A transillumination view by laparoscopy; the view is from laparoscopy
endometrial and scar tissue surrounding the isthmocele is removed. without any light enabling the visualization of the defect with the help
of hysteroscopy light through the defect.
Fig. 6
Laparoscopic tissue removal, a view of the vesicouterine pouch with scar
tissue being pulled and resected with cold scissors.
Fig. 4
Removal of the defect by hysteroscopy; bipolar resectoscopy resects scar
tissue in the isthmocele.
Fig. 7
Laparoscopic suturing of the defect after isthmocele resection suturing
the defect in double-layer suture enabling a thicker and stronger uterine
wall.
Setubal et al. A Review of Uterine Isthmocele Treatment 45
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