Page 4 - Jcog-October 2017
P. 4

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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