Article

A manifestation of endometriosis that will take your breath away: a case report

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Case Report

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American Journal of Emergency Medicine

journal homepage: www. elsevier. com/ locate/ajem

American Journal of Emergency Medicine 33 (2015) 736.e5-736.e7

A manifestation of endometriosis that will take your breath away: a case report

Abstract

Pneumothorax should be included in the differential diagnosis of any patient presenting with sudden onset chest pain or dyspnea, and the di- agnosis should be made promptly and accurately. Catamenial pneumo- thorax, although a rare entity, is a possibility that should be considered in menstruating females presenting with chest pain and/or dyspnea. This case serves to demonstrate the effectiveness of Point-of-care ultrasonography in making the Prompt diagnosis of a pneumothorax, which in this case was a catamenial pneumothorax.

Catamenial pneumothorax (CP) is a rare entity that is often underdiagnosed [2]. It is described as the development of a Spontaneous pneumothorax 48 to 72 hours before or after the onset of menses in women of childbearing age. The pathophysiology remains obscure, and a clear understanding of the mechanism still eludes the scientific community today, although it was first described in 1958 [1].

Point-of-care thoracic ultrasound has been proven to be an effective tool in the emergency setting for multiple applications. This modality has been found to be more specific and sensitive than traditional chest radiography for the diagnosis of pneumothorax [3]. This case clearly demonstrates the advantages of point-of-care ultrasound in the case of a rare presentation of pneumothorax.

A 37-year-old woman with a history of endometriosis and a large endometrioma presented to the emergency department with a chief complaint of chest pain for 1 day. She was seen in the emergency de- partment 3 days before this presentation for abdominal pain and was discharged home to follow up with her gynecologist, after computed to- mography (CT) of the abdomen and pelvis showed no significant or acute findings. On this visit, she states she no longer had any abdominal pain but rather had severe right-sided chest pain and shortness of breath for 1 day. She states the pain began suddenly and that supine po- sitioning exacerbated her symptoms. She denied any fever, nausea, vomiting, or diarrhea. Of note, her last menstrual period began 3 days before this visit. Her vital signs were as follows: pulse, 112 beats per minute; respiratory rate, 30 breaths per minute; and oxygen saturation of 92% on room air. An electrocardiogram revealed uncomplicated sinus tachycardia. The patient was a healthy-appearing young woman in mild respiratory distress, speaking in full clear sentences. Physical examina- tion revealed tachycardia and decreased breath sounds at the right lung base. There was no Lower extremity edema or swelling. Her initial upright posterior-anterior chest radiograph demonstrated a small Right-sided pleural effusion with no other abnormalities (Fig. 1). This was confirmed with the radiology department. A bedside thoracic

ultrasound was performed by an ultrasound fellowship-trained emer- gency physician. Sonography using both B mode and motion

(M) mode of the right anterior chest wall showed no evidence of Lung sliding (Fig. 2a), which raised the suspicion of pneumothorax.

The left lung had no sonographic abnormalities. A CT of the chest was performed to further evaluate the contradictory findings, when so- nography was compared with radiography. The CT revealed a moderate sized right-sided hemopneumothorax (Fig. 3a and b). Given the find- ings and the patient’s symptoms, thoracostomy tube placement was performed using a 24F catheter chest tube. Two hundred milliliters of blood returned from the tube immediately. A follow-up radiograph 1 hour later showed interval resolution of the hemopneumothorax. The patient was admitted to the thoracic surgery service with obstetrics and gynecology consultation. The chest tube was removed on hospital day 3, and the obstetrics and gynecology service recommended begin- ning hormonal suppression therapy. The decision was made to forgo surgical exploration of the pleura and diaphragm, as this was the patient’s first presentation to health care with a hemopneumothorax. The patient’s clinical status improved, and she was discharged home in stable condition on hospital day 5. She was to follow up with obstet- rics and gynecology within the week to schedule elective removal of the endometrioma.

Fig. 1. Initial Chest X-ray.

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Fig. 2. Motion mode ultrasound of right lung showing no lung sliding. Fig. 4. pleural line.

Catamenial pneumothorax is a Rare condition, which exclusively af- fects women during their reproductive years. Although CP has been known to exist for more than a half a century, a clear explanation of its pathophysiology eludes us to this day [4]. It is encountered in 3% to

Fig. 3. CT- scan of Chest.

6% of spontaneous pneumothorax cases in menstruating women, with hemopneumothorax being an even more rare entity accounting for only 14% of those initial 3% to 6% [5]. Maurer et al [1] first described CP in 1958, and his findings suggested a temporal relationship between the onset of pneumothorax and menses, with symptoms typically pre- senting 24 to 36 hours after the onset of menses.

There is no consensus regarding the exact mechanism, but it is thought to involve preexisting or acquired diaphragmatic defects and endometrial implants. Given the relationship between menses and CP, the presence of pelvic endometriosis is of etiologic importance. Pelvic endometriosis is present in 31% to 50% of CP cases [2]. Through further investigation, Maurer et al [1] proposed the main hypothesis to explain this syndrome: a dissolving cervical mucous plug may allow the ascent of air through the fallopian tubes, causing a transient pneumoperitone- um. This Free air would subsequently escape through diaphragmatic de- fects entering into the pleural space, causing CP. The diaphragmatic defects are thought be a result of hormone-regulated sloughing of endo- metrial tissue during menses on the diaphragm itself [2].

On rare occasions, endometrial tissue can be found in the thoracic cavity itself [1,5]. Catamenial pneumothorax is the most common man- ifestation of thoracic endometriosis (73%). Nevertheless, pleural endo- metriosis is found in only half of the patients with CP who were surgically explored [6,7].

The patient in this case patient presented with a hemo- pneumothorax, which suggests that she likely developed some form of thoracic endometriosis, where the endometrial cells bled into her Pleural cavity during menstruation. As with this patient, nearly 90% of CPs occur on the right side [8].

Fig. 5. Normal lung sliding and the “Seashore sign”.

G. Patel et al. / American Journal of Emergency Medicine 33 (2015) 736.e5736.e7 736.e7

Fig. 6. No lung sliding and the “Barcode sign”.

Our patient’s initial chest radiograph did indeed show fluid accumu- lation above the right hemidiaphragm; however, several clinicians missed the subtle findings of a pneumothorax at first glance. Traditional chest radiography has long been the test of choice for the rapid diagno- sis of pneumothorax in the emergency setting. However, it has been widely reported that there are limitations regarding accuracy. Some studies report a 50% miss rate on supine chest radiographs and nearly 30% for Occult pneumothorax on upright AP chest radiographs [9].

With rapid accurate diagnosis being of utmost importance in expe- diting resuscitation, the role of point-of-care thoracic ultrasound has been widely implemented in the emergency setting. The technique in- volves longitudinal scanning of the anterior chest wall with the patient in a supine position. A high-frequency Linear probe is preferred but not mandatory. The probe is placed on the chest between the third and fourth intercostal space [11]. First, the rib acoustic shadows are visual- ized to find the intercostal plane. The pleural line is located as a hyperechoic line visible between and below 2 ribs (Fig. 4). In the normal subject, this pleural line is usually characterized by “lung sliding,” which is a to-and-fro movement of the visceral pleura synchronized with res- piration as visualized in real-time scanning. In the patient with a pneu- mothorax, this finding is typically absent. More so than direct visualization, the use of M mode allows us to more accurately assess pleural sliding or a lack thereof. In normal lung sliding, M-mode imaging demonstrates a linear, laminar pattern in the tissue superficial to the pleural and a granular or “sandy” appearance deep to the pleural line. This phenomenon is known as the “seashore sign [11] (Fig. 5).”

As seen in the images of the right chest of this patient, the linear pat- tern seen in the superficial tissue is also seen below the pleural line. This pattern shows the absence of movement above and below the pleural line suggesting the presence of a pneumothorax. The pattern is known as the “barcode” or “stratosphere sign [11] (Fig. 6).”

Several studies have demonstrated high sensitivity and specificity for thoracic ultrasound in the detection of occult pneumothorax in crit- ical care and trauma patients. The studies of Lichtenstein and Menu [3] involved critically ill patients in an intensive care setting. Absence of lung sliding was found in a prospective operator-blinded study to be a

useful sign for pneumothorax, with sensitivity of 95.3%, specificity of 91.1%, and negative predictive value of 100%. Kirkpatrick et al [10] stud- ied ultrasound vs chest radiography in the trauma patients. Using chest CT as the criterion standard, chest radiography showed a sensitivity of 75.5% (95% confidence interval [CI], 61.7%-86.2%) and a specificity of 100% (95% CI, 97.1%-100%). The sensitivity for ultrasound was 98.1%

(95% CI, 89.9%-99.9%), and the specificity was 99.2% (95% CI, 95.6%- 99.9%). It should be noted that although the absence of lung sliding sug- gests the presence of pneumothorax, other entities such pulmonary fi- brosis, pleural adhesions, acute respiratory distress syndrome, and those who have undergone pleurodesis might also lack lung sliding [10]. Catamenial pneumothorax as an extremely rare condition that is underrecognized and underdiagnosed (needs reference). Although rel- atively little is still known about CP, clinicians should have a high suspi- cion in any female patient of childbearing age with symptoms consistent with pneumothorax. Timely diagnosis and treatment may significantly reduce patient morbidity and mortality; and with the ad- vent of point-of-care ultrasound, clinicians now find themselves with this important tool to aid them in this pursuit. Ultrasound is fast, accu-

rate, and reduces the need for radiation and its cumulative effects.

Gaurav Patel, MD? Brendon Stankard, PA Robert Gekle, MD Steve Park, MD Adam Rucker, MD

Department of Emergency Medicine, North Shore University Hospital

300 Community Drive, Manhasset, NY 11030

?Corresponding author

E-mail address: [email protected]

http://dx.doi.org/10.1016/j.ajem.2014.10.051

References

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