Page 5 - Jcog-October 2017

Reid et al.

J o u r n a l o f C a s e s i n
Obs tetrics & G ynecology

pain, which progressed to include pain in her right shoul- racotomy was proposed in the hope of resecting the sus- Discussion es for symptoms. Both CT and MRI findings have been de-
der and a sensation of shifting fluid in her right thoracic pected foci of endometriosis. After discontinuing leupro- scribed [12], however they are non-specific. Cancer antigen
cavity. In recent years, these cyclic symptoms had wors- lide, an MRI was performed when the patient’s thoracic Thoracic endometriosis is a rare manifestation of 125 (CA 125) has been reported to be elevated in at least one
ened to include shortness of breath and pleuritic chest pain. symptoms recurred. A moderate sized right pneumotho- a common gynecologic condition. Symptoms such case of CP [16], however this too is a non-specific finding.
rax was noted, as well as tiny foci of increased signal as recurrent pneumothorax, dyspnea, and chest pain The optimal treatment for CP is unknown. Hormonal sup-
on the diaphragmatic and anteromedial pleural surface. typically occur within two days of menses [12]. pression alone is associated with high recurrence rates
Figure 1. Thoracoscopy was performed with a gynecologist in at- Multiple etiologic theories have been proposed to explain [8,16]. In one of the largest reviews done to date, Joseph et
tendance. A diaphragmatic lesion was suggestive of endo- the phenomenon of catamenial pneumothorax [13]. The al. [18] suggest that surgery is the superior treatment due to
metriosis (Figure 1). A partial resection of diaphragm and first theory is that during menstruation, partial dilation of the lower recurrence rates. While definitive surgery in the form
talc pleurodesis was performed. The pathology report was cervix and dissolution of cervical mucus allows air to tra- of hysterectomy and bilateral salpingo-oophorectomy is ef-
consistent with full thickness endometrial tissue (Figure 2). verse the uterus and fallopian tubes into the abdomen. From fective [18], these women often require add-back estrogen
Six months post-operatively, the patient continued to expe- here, air passes through diaphragmatic defects. Such fenes- therapy and is unclear whether remaining pleural implants
rience cyclic thoracic pain for two weeks each month, which trations of the diaphragm are more commonly right-sided, would become symptomatic under the effect of exogenous
was satisfactorily managed with anti-inflammatories and she which could account for the predominance of right-sided estrogen. Some small case series have described exclu-
had no further pneumothorax. There had never been any ev- pleural endometriosis [7]. This theory is weakened by ev- sive management with thoracic surgery without subsequent
idence of pelvic endometriosis either based on symptoms or idence that women undergoing insufflation of the abdo- hormonal suppression with good effect [19]. Other authors
clinical examination therefore no laparoscopy was performed. men for laparoscopic surgery do not suffer pneumothorax. argue that the risk associated with thoracic surgery should
The patient subsequently went on to have three However, pelvic instrumentation has been associated with make operative management a last resort [17]. While the
healthy pregnancies. At the time of her three cesare- thoracic endometriosis [14]. A second theory proposes that current consensus regarding treatment is for adjuvant ovari-
an sections, significant pelvic endometriosis was noted. increased prostaglandin F2α levels characteristic of men- an suppression following thoracic surgery [12], there is lack
Gross pathologic specimen of resection portion of struation cause concurrent vasoconstriction and broncho- of robust evidence to support this approach. Moreover, typ-
right hemidiaphragm with multiple endometriosis spasm, leading to ruptured alveoli and resultant pneumotho- ically suppressive medication like danazol and leuprolide
implants with the powder burn appearance classic rax [13]. A third theory proposes that ectopic endometrial are not entirely benign and have undesirable side effects.
for endometriosis pelvis. Figure 2. tissue in the thoracic cavity degenerates in responses to the An important avenue for future research would be evaluat-
ing the efficacy of novel agents (e.g., dienogest) in the treat-
monthly fluctuations in hormones resulting in bleeding into ment of thoracic endometriosis. Certainly, there is compel-
the thoracic space, thereby causing pneumothorax [15].
Following her second pneumothorax in a span of two months, By all accounts, thoracic endometriosis remains underdi- ling evidence that dienogest is a highly effective adjuvant
she underwent a mini-thoracotomy, along with apical bullec- agnosed for several reasons [14]. Cullen Richardson treatment in treatment of pelvic endometriosis [20-22].
tomy and pleurodesis. No endometrial deposits or diaphrag- once said, “The eyes can not see what the mind does not All currently available treatment options offered by gyne-
matic defects were identified at the time of surgery. The final know.” That is, in order for a diagnosis to be made, it cology, whether medical or surgical, are incompatible with
pathology was significant for only a small sub-pleural bulla. must first be considered. Thus, heightened clinical sus- desire for fertility. It is this population, as the woman in our
Following surgery, she continued to experience cyclic picion is required in order to make a diagnosis of thorac- case, who may benefit from a surgical approach targeted at
pleuritic pain, and had a small right apical pneumotho- ic endometriosis. Even the most astute thoracic surgeon removing the offending lesions in the chest. In this instance,
rax that spontaneously resolved. To determine whether may not obtain menstrual history or recognize endometri- video-assisted thoracoscopic surgery is of diagnostic and
her thoracic symptoms were truly related to menstruation, osis implants at the time of thoracoscopic surgery. Sim- therapeutic value and is considered the treatment of choice
a trial of a gonadotropin releasing hormone, leuprolide, ilarly, even a savvy gynecologist is unlikely to include for pneumothorax requiring surgical intervention [12].
was undertaken as a diagnostic test. Menstrual suppres- thoracic symptoms in their typical review of systems for Thoracic endometriosis is a clinical phenomenon that ne-
sion resulted in elimination of her recurrent pneumothorax endometriosis. Moreover, their systematic inspection of cessitates involvement of two medical specialties that
and significant alleviation of chest pain. Over the course the abdominal cavity at the time of diagnostic laparosco- otherwise rarely intersect. As in our case, surgery can
of one year, a total of three courses of oral medroxypro- Same pathologic specimen of right hemidiaphragm py may be too cursory to identify diaphragmatic lesions. be of both diagnostic and therapeutic benefit. Howev-
gesterone acetate (5 mg daily for 10 days) were adminis- in cross section demonstrating a bleb of endometri- Diagnosis of thoracic endometriosis proves challenging as er, the surgeon must be aware of the potential diagnosis
tered to induce withdrawal bleeding and observe for re- osis. there are no non-invasive investigations capable of confirm- in order to detect the often subtle intraoperative findings
turn of symptoms. Onset of bleeding was accompanied ing its presence. Unfortunately, imaging modalities have and refer on to gynecology to discuss the potential clini-
each time by a return of right upper quadrant pain radi- not proven to be sensitive for diagnosis of thoracic endome- cal implications of endometriosis and the potential need
ating to the right shoulder, and lasting about two weeks. triosis and their use is limited to ruling out alternative caus- for ovulatory suppression for further symptom control.
Prolonged ovarian suppression was incompatible with We suggest that all reproductive-aged women presenting
the patient’s desire for pregnancy, therefore a second tho-

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