catamenial pneumothorax caused by endometriosis in the visceral pleura

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as a marker for RV dysfunction in acute pulmonary embo- lism. J Am Coll Cardiol 2002;39:2080. Catamenial Pneumothorax Caused by Endometriosis in the Visceral Pleura Kazuhiro Sakamoto, MD, Takahiro Ohmori, MD, and Hidefumi Takei, MD Department of Respiratory Surgery, Yokohama Rosai Hospital, Yokohama, Japan Catamenial pneumothorax is a rare clinical entity of unknown etiology. The most well known hypothesis is passage of air from the genital tract through endometrial fenestrations in the diaphragm. Although some reports are associated with diaphragmatic endometriosis, few have been confirmed endometrial implants in the vis- ceral pleura. We describe a very rare case of catamenial pneumothorax caused by ectopic endometriosis in the visceral pleura confirmed histopathologically in a woman 1-year after hysterectomy. (Ann Thorac Surg 2003;76:290 –1) © 2003 by The Society of Thoracic Surgeons C atamenial pneumothorax (CP) is a rare disease, and its pathogenesis is poorly understood. Various the- ories have been formulated to explain the development of CP. One of the most widely known theories is that air passes through the genital tract into the thorax through the peritoneal cavity and diaphragmatic fenestrations [1– 4]. We report a case of CP that developed after hysterectomy. A 41-year-old woman had had a hysterectomy for a uterine leiomyoma and monthly abdominal discomfort associated with her menses. Abdominal endometriosis was confirmed at the operation. Postoperatively, the patient still had monthly abdominal discomfort; how- ever, no medication for endometriosis was prescribed. One year after hysterectomy, monthly episodes of chest pain with abdominal discomfort developed. On evalua- tion of the chest pain, she was found to have a small right-sided pneumothorax. She had three documented episodes of right-sided pneumothorax, occurring at about 2-month intervals during a 5-month period. These pneumothoraces were small and treated conservatively. On the third episode of pneumothorax we performed thoracoscopic surgery. A small area of fine adhesion was visualized between the visceral pleura and the chest wall in the right upper lobe of the lung. Several blue-brown spots, less than 3-mm diameter, were found on the tendinous portion of the diaphragm (Fig 1). There were also several dark red dimples, less than 5-mm diameter, on the visceral pleura of the lateral aspect of the upper lobe. No blebs or bullae were visualized. The adhered area of the lung, one of the areas with a small dimple in the upper lobe, and the lesions in the diaphragm were resected with endo- staplers. We widely covered the staple line of the lung and the diaphragm with absorbable mesh sheets. Patho- logically, ectopic endometrial tissue was found in the resected lesions on the diaphragm. The portions of the lung resected from the area with adhesion and the area with dimples also contained endometrial tissue in the visceral pleura (Fig 2). Postoperative recovery was un- eventful, and the patient was discharged on the third postoperative day. She had three additional episodes of recurrent pneu- mothorax associated with chest pain starting 2-months after the operation. After the last episode of recurrent pneumothorax, she elected to start gonadotropin- Accepted for publication Dec 31, 2002. Address reprint requests to Dr Sakamoto, First Department of Surgery, Yokohama City University School of Medicine, 3-9 Fukuura, Kanazawa- ku, Yokohama 236-0004, Japan; e-mail: [email protected]. Fig 1. Thoracoscopic view of several small blue-brown spots (ar- rows) on the tendinous part of the diaphragm. Fig 2. Microphotograph illustrating endometrial tissue in the thick- ened visceral pleura (hematoxylin and eosin stain, magnification 40). 290 CASE REPORT SAKAMOTO ET AL Ann Thorac Surg POST-HYSTERECTOMY CATAMENIAL PNEUMOTHORAX 2003;76:290 –1 © 2003 by The Society of Thoracic Surgeons 0003-4975/03/$30.00 Published by Elsevier Inc PII S0003-4975(03)00155-3 CASE REPORTS

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Page 1: Catamenial pneumothorax caused by endometriosis in the visceral pleura

as a marker for RV dysfunction in acute pulmonary embo-lism. J Am Coll Cardiol 2002;39:2080.

Catamenial Pneumothorax Causedby Endometriosis in the VisceralPleuraKazuhiro Sakamoto, MD, Takahiro Ohmori, MD, andHidefumi Takei, MD

Department of Respiratory Surgery, Yokohama Rosai Hospital,Yokohama, Japan

Catamenial pneumothorax is a rare clinical entity ofunknown etiology. The most well known hypothesis ispassage of air from the genital tract through endometrialfenestrations in the diaphragm. Although some reportsare associated with diaphragmatic endometriosis, fewhave been confirmed endometrial implants in the vis-ceral pleura. We describe a very rare case of catamenialpneumothorax caused by ectopic endometriosis in thevisceral pleura confirmed histopathologically in awoman 1-year after hysterectomy.

(Ann Thorac Surg 2003;76:290–1)© 2003 by The Society of Thoracic Surgeons

Catamenial pneumothorax (CP) is a rare disease, andits pathogenesis is poorly understood. Various the-

ories have been formulated to explain the developmentof CP. One of the most widely known theories is that airpasses through the genital tract into the thorax throughthe peritoneal cavity and diaphragmatic fenestrations[1–4]. We report a case of CP that developed afterhysterectomy.

A 41-year-old woman had had a hysterectomy for auterine leiomyoma and monthly abdominal discomfortassociated with her menses. Abdominal endometriosiswas confirmed at the operation. Postoperatively, thepatient still had monthly abdominal discomfort; how-ever, no medication for endometriosis was prescribed.One year after hysterectomy, monthly episodes of chestpain with abdominal discomfort developed. On evalua-tion of the chest pain, she was found to have a smallright-sided pneumothorax. She had three documentedepisodes of right-sided pneumothorax, occurring atabout 2-month intervals during a 5-month period. Thesepneumothoraces were small and treated conservatively.On the third episode of pneumothorax we performedthoracoscopic surgery.

A small area of fine adhesion was visualized betweenthe visceral pleura and the chest wall in the right upper

lobe of the lung. Several blue-brown spots, less than3-mm diameter, were found on the tendinous portion ofthe diaphragm (Fig 1). There were also several dark reddimples, less than 5-mm diameter, on the visceral pleuraof the lateral aspect of the upper lobe. No blebs or bullaewere visualized. The adhered area of the lung, one of theareas with a small dimple in the upper lobe, and thelesions in the diaphragm were resected with endo-staplers. We widely covered the staple line of the lungand the diaphragm with absorbable mesh sheets. Patho-logically, ectopic endometrial tissue was found in theresected lesions on the diaphragm. The portions of thelung resected from the area with adhesion and the areawith dimples also contained endometrial tissue in thevisceral pleura (Fig 2). Postoperative recovery was un-eventful, and the patient was discharged on the thirdpostoperative day.

She had three additional episodes of recurrent pneu-mothorax associated with chest pain starting 2-monthsafter the operation. After the last episode of recurrentpneumothorax, she elected to start gonadotropin-

Accepted for publication Dec 31, 2002.

Address reprint requests to Dr Sakamoto, First Department of Surgery,Yokohama City University School of Medicine, 3-9 Fukuura, Kanazawa-ku, Yokohama 236-0004, Japan; e-mail: [email protected].

Fig 1. Thoracoscopic view of several small blue-brown spots (ar-rows) on the tendinous part of the diaphragm.

Fig 2. Microphotograph illustrating endometrial tissue in the thick-ened visceral pleura (hematoxylin and eosin stain, magnification�40).

290 CASE REPORT SAKAMOTO ET AL Ann Thorac SurgPOST-HYSTERECTOMY CATAMENIAL PNEUMOTHORAX 2003;76:290–1

© 2003 by The Society of Thoracic Surgeons 0003-4975/03/$30.00Published by Elsevier Inc PII S0003-4975(03)00155-3

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Page 2: Catamenial pneumothorax caused by endometriosis in the visceral pleura

releasing hormone analog (GnRH-analog) therapy. She iscurrently symptom-free after 14 months of follow-up.

Comment

Four theories for the pathogenesis of CP have beenproposed: (1) loss of cervical mucus during the menstrualphase, allowing movement of air into the peritoneum,then the air traverses through the diaphragmatic fenes-trations into the pleural space [1–4]; (2) spontaneousrupture of bullae or blebs during hormonal changes [2];(3) elevated levels of prostaglandin in the serum duringmenstruation, which could cause severe vascular andbronchoalveolar constriction, producing alveolar rupture[2, 4]; and (4) ectopic endometrial tissue in the visceralpleura and its desquamation during menstruation [4, 5].

Given that our patient had previously had a hysterec-tomy, it is unlikely that the CP was caused by movementof air through the cervix. In our patient, no blebs orbullae were found after careful thoracoscopic inspectionof the lung. Thus, our report supports the theory that CPis caused by endometrial implants, which could havetraveled across diaphragmatic fenestrations or lymphaticchannels to reach the visceral pleura [2, 3]. A commontheory regarding the cause of endometriosis is that viableendometrial cells can reflux through the fallopian tubesduring menstruation, implant, and grow on the sur-rounding pelvic structures and peritoneum [6]. Thesecells could implant in the visceral pleura through thefenestrations in the diaphragm; however, pathologicallyconfirmed endometriosis in the visceral pleura is ex-tremely rare in the literature [5]. Our case confirms thatvisceral pleural endometriosis can be a cause for CP.Previous case reports of recurrent CP after diaphrag-matic resection could theoretically be due to visceralendometrial implants. Therefore, careful inspection ofthe entire lung surface at the time of operation for CP ismandatory and systemic therapy with GnRH-analogs [2,4], pleurodesis, or both should be considered on thetreatment for CP.

We thank Shunji Kawamura, MD, for pathologic support.

References

1. Lillington GA, Mitchell SP, Wood GA. Catamenial pneumo-thorax. JAMA 1972;219:1328–32.

2. Van Schil PE, Vercauteren SR, Vermeire PA, Nackaerts YH,Van Marck EA. Catamenial pneumothorax caused by thoracicendometriosis. Ann Thorac Surg 1996;62:585–6.

3. Cowl CT, Dunn WF, Deschamps C. Visualization of diaphrag-matic fenestration associated with catamenial pneumothorax.Ann Thorac Surg 1999;68:1413–4.

4. Blanco S, Hernando F, Gomez A, Gonzalez MJ, Torres AJ,Balibrea JL. Catamenial pneumothorax caused by diaphrag-matic endometriosis. J Thorac Cardiovasc Surg 1998;116:179–80.

5. Flieder DB, Moran CA, Travis WD, Koss MN, Mark EJ.Pleuro-pulmonary endometriosis and pulmonary ectopic de-ciduosis: a clinicopathologic and immunohistochemical studyof 10 cases with emphasis on diagnostic pitfalls. Hum Pathol1998;29:1495–503.

6. Rock JA, Markham SM. Pathogenesis of endometriosis. Lan-cet 1992;340:1264–7.

Liver Metastases From LungCancer: Is Surgical ResectionJustified?Isidoro Di Carlo, MD, PhD, Giuseppe Grasso, MD,Domenico Patane’, MD, Domenico Russello, MD, andFerdinando Latteri, MD

Department of Surgical Sciences, Organs Transplantation andAdvanced Technologies, University of Catania, andDepartments of Pathology and Radiology, CannizzaroHospital, Catania, Italy

Resection of the liver for metastatic lesions has largelybeen done for secondary colorectal or neuroendocrinetumors, and there is little information of its value forother lesions. Recent improvements in hepatic surgeryhave made resection of metastases a safe procedure and itshould certainly be considered whenever there is anisolated lesion. We report the case of a successful resec-tion of an isolated secondary hepatic lesion from a lungprimary tumor, which was resected approximately 4years beforehand. A review of the literature demon-strates that although early reports of similar procedureswere not favorable, more recent reports reinforce thevalue of an aggressive approach in favorable cases.

(Ann Thorac Surg 2003;76:291–3)© 2003 by The Society of Thoracic Surgeons

Liver resections for metastases are commonly per-formed for colorectal primary tumors and are poorly

documented for noncolorectal tumors. Several authorssuggest that liver resection of noncolorectal secondaries,unless from Wilms tumor, should be discouraged [1],whereas others take the view that selected patients arecandidates for hepatic resection [2]. Few cases of resectedliver metastases from lung cancer are present in theliterature. The aim of this study is to report a patientoperated on for isolated liver metastasis from lung cancerand review the literature.

A 69-year-old woman had a lobectomy for an adenocar-cinoma of the lung in June 1995, which staged well. Fouryears later at routine follow-up, she was found to have a5-cm lesion in the liver located in segment VII (Fig 1). Thepatient remained in good health and neither a gastros-copy or colonoscopy showed evidence of an occult pri-mary. Total body scintigraphy demonstrated no other

Accepted for publication Dec 23, 2002.

Address reprint requests to Prof Di Carlo, Department of SurgicalSciences, Organs Transplantation and Advanced Technologies, Univer-sity of Catania, Cannizzaro Hospital, Via Messina, Catania 95126, Italy;e-mail: [email protected].

291Ann Thorac Surg CASE REPORT DI CARLO ET AL2003;76:291–3 RESECTED LIVER METASTASES FROM LUNG CANCER

© 2003 by The Society of Thoracic Surgeons 0003-4975/03/$30.00Published by Elsevier Inc PII S0003-4975(03)00149-8

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