A Little History

Not something new.

The occurrence of endometriosis cells in tissues of the respiratory system is not something new.  In fact, in the 1880’s vague discussions of endometriosis and the respiratory system were reported.   The first post-mortem diagnosis of decidua tissue in the lungs of a 72 yo female at autopsy in 1912 (1).  The woman had undergone hysterectomy at age 50 yrs.  Multiple subpleural nodules of tissue like that of the uterus were reported  But there are only a few published case studies suggestive of intra-thoracic disease through the 1930’s.  In 1938 Otto Schwartz is reported a case of endometriosis of the inguinal region in a patient with a ‘lung tumor’ which bled with each monthly menses (2).  He speculated that this ‘lung tumor’ was endometriosis. His theory was supported that bleeding ceased after the woman received radiation to the ovaries. Schwartz was unable to obtain histological confirmation of the lesion. During this time frame the medical establishment significantly opposed the possibility that endometriosis could be found in the chest cavity.

In 1940 Hobbs and Bortnick (3) postulated that endometriosis cells could be found within the lung tissue of human females.  To challenge the hypothesis, they injected endometriosis cells into the ear veins of (12) rabbits.   About 10 weeks after injection, necropsy on each rabbit was performed.  Components of glands and/or stroma was identified in the lung tissue of 8/12 rabbits (75%).

The association between the onset of menses and occurrence of a spontaneous pneumothorax in a female was given credit to Elmer Maurer and his coauthors (3).  Maurer presented a case of recurrent spontaneous pneumothorax of the right lung between 1952 and 1954. Resection of the central tendon in the right hemi-diaphragm revealed a 4 cm x 3 cm lesion. W.Wallace Park reported a case of a 26 yo female who died four hours after delivery of her second child (5).  The woman succumbed to pneumonia.  In addition to abscess and empyema, stains of the tissue samples suggested decidua origin:

…The pathological changes are a fibrinous pleural exudate, a zone of inflammatory-cell infiltration with intense vascular congestion extending inwards for 1mm.                                                                                                                – W. Wallace Park (1954)

(Decidua: The thick layer of modified mucous membrane which lines the uterus during pregnancy and is shed with the afterbirth – Oxford Dictionary)

A year following this report, Hartz (6) presented another case with a 24 yo female who died in the 4 month of gestation during hospitalization for hemoptysis (coughing up blood). In addition to findings for congestive heart failure, findings suggestive of pulmonary infarction were reported.  Unique additional findings included:

…the pleura of the lower lobe of the right lung showed fibrous deposit, the color of the lungs on section was brown; there were many foci of condensation and numerous small hemorrhages…

It was obvious at once that the tissue in question did not resemble anything which I had previously observed in normal or diseased lungs; on the other hand, there was strong resemblance to decidua.                                                             – Hartz (1956)

In 1959 Fleishman and Davidson (7) reported a case of recurrent hemoptysis at menses a year following a Dilation and Curettage, and three years after a cesarean section delivery of her second child.  She continued to have intermittent ‘vicarious’ hemoptysis with menses over four years between trial use of testosterone.  Remarkably, radiograph images (x-rays) were able to capture lesions within the lung during a few active episodes concurrent to menses postulating that endometriosis was the cause. The authors concluded that a history of C-Section and D&C procedures, in both, their case and one presented in 1956 by Latters et al (8),  both underwent C-Section and D&C procedures prior to the onset of hemoptysis. They postulated these procedures may have precipitated emboli to the lungs. However, it is important to note that females without a history of uterine manipulation and/or are nulliparous, also develop hemoptysis (9).

In the 1950’s and 1960’s a few published autopsies of women who died during pregnancy or immediate post-partem (6,10-12) Endometriosis ‘nests’ were observed in the lung parenchyma and parietal pleural (chest wall) .  The authors postulated the endometriosis lesions a) arrived through blood vessels (6,10-12) or, b) possibly differentiated from local tissue (8) or displaced embryonic tissue (10,11).

After the the last post-mortem study of 1968 was published, a few small subject number studies were released.  with a few case reports and a limited number of published autopsies of women who died during or following childbirth. Kovarik and Giles (12) described ten cases (6 lung parenchyma and 4 pleura).  However, Rosemary Davies (13) observations of nine cases, concluded that –

“The relationship between the development of the pneumothorax and the onset of menstruation is, in every case, a very close one, the respiratory symptoms occurring either in 24 hours before the onset of menstruation or during the first two days of the period.                                                                              – Rosemary Davies (1968)

In 1973, Lillington et al (14) identified the occurrence of a spontaneous pneumothorax which occurred within 72 hours prior to, and-up-to 72 hours after the onset of menses as ‘Catamenial Pneumothorax.’

The word ‘catamenial’ is derived from the Greek word ‘Katamenios’ meaning monthly occurrence.                                                       – Aikaterini N et al. (2014)

Catamenial Pneumothorax – Review Article. J. Thorac Dis.2014;6(S4):S448-S460.

At a later date, the most commonly used time-period defining ‘catamenial’ was reduced to 24 hours prior-to, through 72 hours after onset of menses (15). This aligns with observations of Davies (1968)

The recognition that endometriosis of the respiratory system was, in-fact, responsible for lung collapses (pneumothorax), bloody pleural effusions (hemothorax) and coughing up blood (hemoptysis), (as well as lung nodules identified on radiographs or intra-operatively) became known as: Thoracic Endometriosis Syndrome (TES) (16).

Over the past few decades, investigation has led to the understanding that TES is not ‘rare’. In fact, the manifestations of disease may occur, in lesser frequency, outside of the ‘catamenial’ time frame.  Prior to 2015, pneumothorax which occurred due to endometriosis outside of the catamenial time period were referred to as: ‘Non-Catamenial Endometriosis Related’. Fukuoka et al (17) established the term: Thoracic Endometriosis Related Pneumothorax (TERP).  TERP identifies ALL spontaneous pneumothorax that occur outside of the ‘catamenial’ time period related to endometriosis. TERP is estimated to result in 8-10%, and Catamenial Pneumothoraxes are estimated in 24-25% of all females adolescent to perimenopause due to endometriosis (18).

                                      Citations: ‘Not Something New’                                         TEReferencesNotSomethingNew07192018



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