Keywords
deep infiltrating endometriosis - intestinal endometriosis - intestinal rupture -
pregnancy complications
Palavras-Chave
endometriose infiltrativa profunda - endometriose intestinal - rotura intestinal -
complicações na gravidez
Introduction
Endometriosis is a progressive and benign estrogen-dependent disease defined by the
presence of endometrial tissue (glands and stroma) outside the uterine cavity. Deep
infiltrating endometriosis (DIE) is considered a specific entity, which has been arbitrarily
defined in histological terms as endometriotic lesions extending more than 5 mm underneath
the peritoneum, and it is responsible for painful symptoms, with some women experiencing
severe symptoms, while others remain asymptomatic.[1] Due to the variable clinical presentation, the lack of pathognomonic symptoms, and
the fact that no useful noninvasive clinical tests to diagnose the symptomatic disease
are available, a delay in the diagnosis that averages from 5 to 11 years is observed.[1]
[2]
Deep infiltrating endometriosis is found in 20% of women with endometriosis. Bowel
involvement is diagnosed in 5% to 12% of patients with endometriosis, with most lesions
(90%) located in the colorectum. The role of colorectal endometriosis in women with
infertility remains to be established.[1]
[3] Unfortunately, in most published studies, fertility and pregnancy data are underreported
or not fully considered.[4] Intestinal DIE is a severe disease that may affect young women desiring pregnancy.
Digestive symptoms may be found in association with deep dyspareunia, infertility,
and impaired quality of life. In order to determine the best approach to bowel endometriosis,
several factors such as clinical symptoms, extent of the disease and imaging evaluation
results should be taken into consideration.[2]
[4]
[5]
Case Summary
We report the case of a 33 year-old woman who complained of severe dysmenorrhea since
menarche. From 2003 to 2009, she underwent 4 laparoscopies for the treatment of pain
associated with endometriosis (dysmenorrhea, dyschezia). The first surgery, which
was performed in 2003, revealed endometriosis grade IV. A gonadotropin-releasing hormone
(GnRH) analogue (goserelin) was administered to her for 6 months after surgery, but
the pain recurred soon after her menses returned. As the pelvic pain persisted, a
new laparoscopy was performed in 2006, and the levonorgestrel intrauterine system
(LNG-IUS, Mirena, Bayer, Leverkusen, Germany) was inserted, with partial pain relief.
In 2008, she started complaining of dyschezia, and underwent a third laparoscopy,
for which no records are available. In spite of using Mirena, the pain, as well as
the dyschezia, persisted. Thus, she underwent a fourth surgery in 2009. Finally, a
colonoscopy performed in 2010 revealed rectosigmoid stenosis probably due to extrinsic
compression. She was advised to get pregnant before attempting to treat the intestinal
lesion, in view of the risks involved in such a surgery.
Spontaneous pregnancy occurred soon after the LNG-IUS was removed in 2011. When the
patient was in the 33rd week of pregnancy, she started feeling severe abdominal pain.
No fever or sings of pelviperitonitis were present, but as the pain worsened, a cesarean
section was performed, with the delivery of a premature healthy male. Unfortunately,
an intestinal rupture was also identified. Severe peritoneal infection and sepsis
ensued. A colostomy was performed, and the patient recovered after 8 days in intensive
care. Three months later, the colostomy was closed, and a new Mirena was inserted.
Only then did the patient come for treatment of the intestinal endometriosis with
our multidisciplinary endometriosis team. The diagnostic evaluation, including a transvaginal
ultrasound (TVUS) with intestinal preparation and a magnetic resonance imaging (MRI)
scan ([Fig. 1]), revealed the presence of intestinal lesions, while the colonoscopy confirmed stenosis
due to extrinsic compression of the rectum ([Fig. 2]).
Fig. 1 Magnetic resonance imaging scan showing an endometriotic nodule in the rectosigmoid.
Fig. 2 Colonoscopy performed before surgery in 2014 showing extrinsic compression in the
rectum by an endometriotic nodule.
The patient then underwent a laparoscopic excision of the endometriotic lesions, including
an ovarian endometrioma, adhesiolysis and segmental colectomy on November 2014. She
is now fully recovered and planning a new pregnancy. A TVUS performed six months after
surgery showed signs of pelvic adhesions, but no endometriotic lesions.
Discussion
Intestinal evaluation is extremely important for the surgical planning, since the
number of lesions and the depth of invasion influence the composition of the surgical
team, the equipment used and the technique chosen. Bowel involvement is frequently
multifocal, and the most commonly affected areas are the rectosigmoid colon, the appendix,
the cecum and the distal ileum.[1]
[5] Although the rectal endoscopic sonographic approach is the most precise approach
for the evaluation of the involvement of intestinal layers, such identification is
also possible by TVUS.[4]
[5] It has been shown that lesions that affect more than 40% of the bowel circumference
reach beyond the inner muscular layer.[3]
[4]
[5]
Women presenting with intestinal symptoms and those with previous endometriosis surgery
are at increased risk of bowel resection.[4]
[5] Our patient presented with recurrent symptoms, including dyschezia, despite four
laparoscopies and the continuous use of medical treatments. The decision to perform
surgery for DIE is mainly clinical.[4]
[5] The TVUS and other imaging techniques such as the MRI can be useful tools to make
a preoperative estimate of the size and lateral extension of the lesions, and they
play a vital role in the surgical planning and approach. It remains unclear, however,
to what extent the preoperative ultrasonography or the MRI should influence the decision
to perform the surgery, or the decision regarding the type of intervention to undertake
for DIE.[5]
[6] According to the European Society of Human Reproduction and Embryology (ESHRE) Guideline,[2] the ureter, the bladder and the bowels should be assessed if DIE is suspected, in
order to establish the extent of the disease. Even though the colonoscopy performed
in 2010 revealed rectosigmoid stenosis probably due to extrinsic compression, the
patient was advised to get pregnant before attempting to treat the intestinal lesion,
in view of the risks involved in such a surgery. As the patient only came to our multidisciplinary
team after intestinal rupture in 2013, we do not know why she was not operated earlier,
even though the decision to perform surgery may vary according to the literature.
Although the accuracy of the colonoscopy for the identification of intestinal involvement
in DIE is debatable[7] in view of the rarity of mucosal involvement, it was indicated in our patient due
to the persistent intestinal symptoms. Indeed, the colonoscopy showed stenosis due
to extrinsic compression. Therefore, surgery should have been contemplated before
pregnancy, even though there is no consensus in the medical literature as to what
is the best approach in such clinical settings.[3]
[6] Once again, she was not previously under our care, and no record of clinical decisions
was available to us.
Surgery is considered the treatment of choice for symptomatic DIE, as the complete
removal of lesions results in significant pain relief and improvement in quality of
life scores.[2]
[4]
[5] The best therapeutic approach for women with DIE involving the sigmoid and/or rectum
remains to be established.[4]
[5] A variety of factors such as clinical symptoms, lesion location and results from
imaging studies (TVUS and MRI), as well as the recurrence rates and impact on fertility
and quality of life should be taken into consideration.[5]
[6] Some advocate that a bowel resection is rarely justified, and that in many cases
involving the rectovaginal septum or the bowel, DIE can be appropriately managed without
the surgical excision of such lesions.[8] A conservative approach seems justified in younger patients wishing to conceive,
although segmental bowel resection and anastomosis result in better outcomes, without
interfering in pregnancy rates.[5] This may have been the case when our patient was first operated, as she was not
infertile. Her clinical history, however, showed intestinal symptoms (dyschezia),
and the medical treatment she underwent apparently was not enough to stop the progression
of her intestinal endometriosis. In addition, the colonoscopy had already showed stenosis
probably related to extrinsic compression. Unfortunately, none of the previous surgeries
were performed by a multidisciplinary endometriosis team. It is advisable to refer
women with DIE to a center of expertise in endometriosis that can offer multidisciplinary
management, as the surgery is associated with significant complication rates.[2]
Disastrously, intestinal rupture during pregnancy occurred. Although rare, intestinal
obstruction due to endometriosis during pregnancy may happen, with rather severe complications.
Unfortunately, DIE might be overlooked, as symptoms can be elusive and unspecific,
and intestinal lesions may be missed even during laparoscopy. Furthermore, smaller
deep lesions, especially at the level of the sigmoid, may be missed during the diagnostic
workup and laparoscopy. Last, but not least, one expects endometriosis to regress
during pregnancy.[2]
[9] Regrettably, there may be more cases, as many may not have been published. We have
received personal communication of two other cases of intestinal rupture after controlled
ovarian stimulation for in vitro fertilization (IVF). As a matter of fact, acute complications
of endometriosis occurring during pregnancy remain rare. Setúbal et al[9] published a review on intestinal complications caused by DIE either during pregnancy
or IVF. Their literature search revealed 12 articles describing 12 complications related
to the progression of DIE during pregnancy, and 1 article reporting 6 cases of bowel
occlusion during IVF. Surgery is not mandatory in all cases, and when infertility
is involved, IVF appears to be the best option.[2]
Women presenting with intestinal symptoms and those with previous endometriosis surgery
are at increased risk of bowel resection. The decision to perform surgery in this
setting, however, remains mainly clinical. Available published data on the long-term
outcomes reveals a cumulative probability of pain recurrence between 20% and 40%,
and need of another surgical procedure between 15% and 20%. Therefore, the decision
to perform radical or conservative surgery or no surgical intervention at all should
be evaluated individually.[10] Accurate information on the benefits and risks involved in the procedure versus
treatment without bowel resection should be available to all patients. The management
of women with bowel endometriosis remains a challenge due to the lack of published
studies evaluating the real risk of symptom recurrence, the effects on fertility,
and the need for further surgical interventions. Decisions, however, should be individualized
to the needs of each woman after providing information on the potential benefits,
harms, and costs of each treatment alternative, as well as after evaluating the presence
of pelvic pain, the woman's age, lesion location, and previous treatments.[10] In such a setting, the evaluation by a multidisciplinary endometriosis team is a
fundamental step in order to achieve successful results.
