Document Type : Case Report
Authors
1 Endometriosis Research Center, Iran University of Medical Science, Tehran, Iran;Department of Obstetrics and Gynecology, Zabol University of Medical Science, Zabol, Iran
2 Endometriosis Research Center, Iran University of Medical Science, Tehran, Iran
3 Endometriosis Research Center, Iran University of Medical Science, Tehran, Iran;Department of Obstetrics and Gynecology, Kerman University of Medical Science, Kerman, Iran
4 4Pain Research Center, Iran University of Medical Science, Tehran, Iran;5Department of Anesthesiology, Zabol University of Medical Science, Zabol, Iran
Abstract
Keywords
Endometriosis, which is characterized by the incidence of
stroma and endometrial glands outside of the uterine cavity, is
common in approximately 10% of women during their child
bearing age (
Here in, we present a case of diaphragmatic endometriosis associated with pelvic endometriosis in a 20-year-old female patient with chronic pelvic pain and dysmenorrhea with a high score. In a preliminary investigation, she was diagnosed with deep pelvic endometriosis. However, during laparoscopic surgery of the entire abdominal and pelvic cavity, diaphragmatic endometriosis was discovered incidentally, which had spread through the center and right parts of diaphragm. In this case report, we introduce a rare case of diaphragmatic endometriosis along with pelvic endometriosis and discuss its symptoms and therapeutic methods.
In March of 2017, a 20-year-old virgin female with achronic pelvic pain was referred to our center. The patient complained of severe pelvic pain with verbal numerical rating scale (VNRS) of 9 during the menstrual cycle. This chronic pain had lasted for almost one year. The patient did not mention dyschezia, pain during or afterurination, orother symptoms associated with diaphragmatic endometriosis, such as chest pain, shoulder pain, or right upper abdominal pain. Furthermore, she had used no hormone replacement therapy.
In abdominal examination, there was fullness on the left side, while in both rectal examination and abdominal examination, there was fullness in the posterior cul-de- sac. An immobile 10-cm mass wasfelt on the left side, whereas another immobile 5-6-cm mass was on the right side that was fixed to the uterus.
Pelvic ultrasonography results indicated a cyst with an approximate size of 12×7 cm consisting of thick contents in the left ovary with internal septae, raising suspicion regarding formation of the tubo ovarian complex in endometrial cavity. Furthermore, the ultrasound findings showed an endometrium a cyst with an approximate dimension of 4 cm on the right side with adhesion and endometrial nodule of the posterior fundus with moderate adhesion to the rectosigmoid. Therefore, magnetic resonance imaging (MRI) was performed to exclude the left mass from adenocarcinoma, while the results showed normal upper abdominal organs, including liver, spleen, pancreas, kidneys, adrenal, as well asthe lungs. In pelvic MRI findings, there was endometrium in both adnexae along with hydrosalpinx on the left side, whereas enhancement was not reported in the left adnexal masses.
In addition, the blood test showed an anti mullerian hormone (AMH) of 1.82 and CA-125 of 125.1, while other tumor markers, including risk of ovarian malignancy algorithm (ROMA) and HE4 were normal.
During laparoscopy, we noticed extensive endometriosis that involved the anterior and posteriorcul-de-sac, both pelvic side walls, both ovaries, and sigmoid colon. The left ovary contained a cyst measured 10-12 cm with severe adhesion to the rectum, while the right ovary contained a cyst measured approximately 6 cm with moderate adhesion to the tube and the right ovary. There was also no evidence of endometriosis in ureters. Anatomy of pelvis restored, pelvic Die corrected and a 2-cm endometriotic nodule attached to the rectovaginal septum (RVS) was shaved.
On exploring the upper abdomen, 5 to 6 areas of superficial
endometriosis were discovered in the, anterior and center
of the right hemi-diaphragm (Figes
Lesions of endometriosis on the rightside and the center of the diaphragm.
Lesion of endometriosis on the surface on right hemi diaphragm.
It has been reported that the incidence of endometriosis
among the women of child bearing age is about 10%
(
Diaphragmatic endometriosis is a rare serious disorder,
which has been reported for the first time as a separate
term by Brews (
The pain in diaphragmatic endometriosis is due to
stimulation of a sensory branch of the C5 nerve root. The
severity of the symptoms varies depending on the location
and depth of the lesions.It has been reported that
diaphragmatic endometriosis can be asymptomatic, while
some women may experience no clinical symptoms or an
obscure pain (
Diaphragmatic endometriosis isdiagnostic error due to
the similarity of clinical symptoms with other benign or
malignant disorders. About 95% of diaphragmatic lesions
occur in the right side of the diaphragm, although it has
been previously seen in the left side alone or both sides
of the diaphragm, even in some vital structures, like the
phrenic nerve. Furthermore, in most of the reported cases,
the lesions occur in the anterior or posterior portion of the
diaphragm and behind the liver. Therefore, due to the diversity
of an organ site involvement, diaphragms and their
surrounding areas should be thoroughly examined (
In terms of macroscopic appearance, lesions may appear
in different colours and shapes that are mostly reported
as bruised, purple and purple red. Computerized
tomography (CT) scan or MRI may play an important
role in diagnosis. Thoracic endometriosis may appear as
small cystic lesions in chest radiography or CT scan (
Therapeutic measures for diaphragmatic endometriosis or
suspicious thoracic endometriosis may be mainly based on
the patient’s medical history. It has been strongly indicated
that the best treatment choice is the expectant approach as
compared to the other interventions for those patients with
asymptomatic diaphragmatic endometriosis (
However, for symptomatic patients, surgery will be beneficial,
if the medicationis deemed to have failed (
The patient’s age, the type of treatment andthe medication
as well as the surgeon’s expertise should be also considered
in this regard. Although there is still uncertainty
about the efficiency of laparoscopic surgery in diagnosis
and treatment of diaphragmatic endometriosis (
The use of hormonal medications, such as danazol (oral
contraceptives), has been suggested to the patients who
are not interested in VATS or believe the thoracoscopy is
not safe enough. The segmental resection is needed during
VATS for the following disorders: tension pneumothorax,
hemopneumothorax, lesions of pulmonary endometriosis,
chemical pleurodesis, as well as pleurectomy (
In our case, diaphragmatic endometriosis was discovered after inspection of the upper abdomen. In addition, the patient had nosymptoms, such as shortness of breath, shoulder pain, and right upper quadrant (RUQ) pain. Therefore, due to laparoscopic examination of the diaphragm, and appearance of lesions, the endometriosis lesions ablate. We decided to apply no other interventions for the patient.
It has been reported that the asymptomatic diaphragmatic
endometriosis can be safely treated with use of laparoscopic
surgery instead of laparotomy, diaphragmatic
resection, or other interventions (
After discharge, considering the virginity, we prescribed suppressive hormonal medicationsfor the patient. Furthermore, we advised her to go to a hospital immediately if she experience shoulder or RUQ pain, shortness of breath and other catamenial symptoms.
Endometriosis is considered as a clinical puzzle for both physicians and patients. Although many efforts have been made for both diagnosis and treatment of this disease, it is still controversial in terms of clinical symptoms, pathophysiology, disease progression as well as management. The surgeon should be fully aware of the clinical symptoms, patient’s medical history, and endometriosis lesions during a laparoscopic surgery. It is noted that if chest pain, shoulder pain, hemothorax, pneumothorax, and hemoptysis occur during the reproductive age, especially with acyclic pattern, then diaphragmatic endometriosis should be considered. Therefore, a close inspection of both anterior and posterior parts of hemi-diaphragm and applying a combined VATS/laparoscopy procedure are needed. This is especially true if there are lesions present. In order to achieve better outcomes with prevention of recurrence and re-occurring clinical symptoms, resection of any suspicious lesion is also recommended.
It is noteworthy that in contrast to conservative treatment which will be applied when endometriosis is detected during laparoscopic surgery in asymptomatic patients, however, an interventional approach is needed in symptomatic patients with post-surgical complications.