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Ann Thorac Surg 2001;72:1750-1751
© 2001 The Society of Thoracic Surgeons


Case report

Catamenial hemoptysis

Fred Weber, MD, JD*a

a Department of Thoracic Surgery, Shore Memorial Hospital, Somers Point, New Jersey, USA

Accepted for publication February 14, 2001.

* Address reprint requests to Dr Weber, 10 W Connecticut Ave, Somers Point, NJ 08244-0111, USA
e-mail: weber{at}law.com


    Abstract
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 Abstract
 Introduction
 Comment
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Catamenial hemoptysis is a rare result of thoracic endometriosis. The long-term treatment of this condition remains controversial. An operation to remove the offending parenchymal endometrial tissue by pulmonary sparing, such as wedge resection, segmentectomy affords satisfactory treatment without the disadvantage of long-term hormone therapy and with the consequent suppression of normal menses and alteration of secondary sexual characteristics in a young woman.


    Introduction
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 Abstract
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 References
 
Several clinical syndromes associated with the female menses have been described. The first was increased seizure activity in relationship to the menstrual cycle, which was reported by Gowers [1] in 1885. The syndrome of recurrent pneumothorax in relation to menses was initially reported by Mauer and collegues in 1958 [2]. This was labeled catamenial pneumothorax by Lillington and colleagues in 1972 [3]. As of 1991, over 100 cases of catamenial pneumothorax had been reported in the literature [4].

Joseph and Sahn [5] reviewed the English medical literature for cases involving menses and pulmonary pathology. For unification, Joseph coined the term thoracic endometriosis syndrome. Thoracic Endometriosis Syndrome includes the syndromes of catamenial pneumothorax, catamenial hemothorax, catamenial hemoptysis, lung nodules, isolated catamenial chest pain, and catamenial pneumomediastinum. Catamenial hemoptysis is uncommon, with only isolated cases reported in literature searches. A recent review by Terada and colleagues [6] has identified only 30 reported patients with catamenial hemoptysis in the English literature.

Catamenial hemoptysis is a syndrome characterized by bleeding from the bronchial tree and lung that occurs synchronously with the female menstrual cycle. Because this is a much rarer phenomenon than catamenial pneumothorax, there have been no individual series reported and only anecdotal case reports have appeared in the literature. Controversy exists as to the proper treatment for catamenial hemoptysis because it has been treated with both hormone therapy and pulmonary resection. Because of the uncommon nature of the problem and the debate with regard to appropriate therapy, a successfully treated case with long-term follow-up is presented.

A 27-year-old woman was referred because of an unusual pattern of hemoptysis. Over the previous 2 years, on the first day of her menstrual period, she coughed 100 to 200 mL of bright red blood. This continued each month until the time of evaluation. A computerized axial tomography scan of the chest did not reveal any pulmonary abnormalities. A random bronchoscopy was normal and did not reveal the source of the blood.

Because of the lack of a target in the chest, the patient was brought to bronchoscopy when actively bleeding. Fresh blood was seen in the right upper lobe bronchus. Options of treatment including hormonal therapy and surgical resection were discussed with the patient. Because of the disadvantages of hormonal therapy interfering with her ability to bear children, the patient opted for definitive surgical treatment.

On the basis of information obtained at bronchoscopy, a right upper lobectomy was performed. The operative and postoperative course was normal. The pathology specimen revealed old hemorrhage, but 71 blocks of tissue failed to demonstrate any evidence of endometriosis. She is healthy 8 years later and has not had any recurrence of hemoptysis. During this interval, normal gestation and delivery was accomplished without incident.


    Comment
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 Abstract
 Introduction
 Comment
 References
 
The proper treatment of catamenial hemoptysis remains controversial. Methods of treatment center on suppression of the endometrial tissue with hormones and hormone analogues versus surgical extirpation of the endometrial tissue. Because of the anecdotal nature of cases, there are no large series to study in which to base therapy on.

The first case of catamenial hemoptysis with successful treatment was reported in 1956 by Lattes [7]. The patient was treated initially with hormonal manipulation with testosterone, and when the hemoptysis recurred the patient was treated with surgical resection.

Sporadic reports of successful treatment with an operation followed. In 1962, Rodman and Jones reported a patient with bronchial endometriosis resulting in catamenial hemoptysis, which was treated with left apical posterior segmentectomy [8].

More recently, medical therapy similar to the treatment of pelvic endometriosis has been attempted for control of catamenial hemoptysis. This treatment involves suppressing the endometrial tissue with pseudopregnancy induced by progesterone therapy or pseudomenopause achieved with danazol administration. Danazol is a steroid hormone that affects the ovarian hormone synthesis and competitively binds to cytoplasmic receptors for sex steroids in target tissues [9]. The first report of treatment of catamenial hemoptysis with hormones rather than an operation was noted by Ronberg [10] in 1981. Since then danazol has been used successfully in the treatment of catamenial hemoptysis [11].

However danazol therapy is not a cure and when the hormone therapy is discontinued, hemoptysis may recur necessitating lobectomy [12]. In the Joseph and Sahn [5] review, permanent freedom from hemoptysis was achieved in only 3 of the 5 patients placed on hormone therapy.

In addition to the problem of recurrence, hormonal therapy has many side effects that can make it an unattractive therapy for the young women that comprise the patient population. These women are in childbearing years, and the suppression of ovulation and hormonal manipulation precludes pregnancy and childbirth. Secondary sexual characteristics regress in hormonal-treated women. In addition, there is weight gain and change in body habitus. These changes may be unacceptable to a young woman facing long-term hormonal therapy.

By contrast, an operation offers the certainty of being cured without the long-term problems associated with hormone therapy. All efforts must be made to precisely locate the source of bleeding. Although computed tomographic scans were not performed in the reported patients a computed tomography exam during an acute bleed may ultimately be necessary to accurately locate the abnormal area. Then, with exact anatomic localization of the offending area, a tissue-sparing procedure can be planned. The proper treatment for catamenial hemoptysis is a limited pulmonary resection with the removal of all ectopic endometrial tissue. This can be accomplished with a lung-sparing segmentectomy or wedge resection.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Rodriguez Macias K.A. Catamenial epilepsy: Gynecological and hormonal implications. Five case reports. Gynecol Endocrinol 1996;10:139-142.[Medline]
  2. Mauer E.R., Schall J.A., Mendez F.L. Chronic recurring spontaneous pneumothorax due to endometriosis of the diaphragm. JAMA 1958;168:2013-2014.
  3. Lillington G.A., Mitchell S.P., Wood G.A. Catamenial pneumothorax. JAMA 1972;219:1328-1332.[Abstract/Free Full Text]
  4. Shiraishi T. Catamenial pneumothorax: report of a case and a review of the Japanese and non-Japanese literature. Thorac Cardiovasc Surg 1991;39:304-307.[Medline]
  5. Joseph J., Sahn S. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996;100:164-169.[Medline]
  6. Terada Y., Chen F., Shoji T., Itoh H., Wada H., Hitomi S. A case of endobronchial endometriosis treated by subsegmentectomy. Chest 1999;115:1475-1478.[Abstract/Free Full Text]
  7. Lattes R., Shepard F., Tovell H., Wylie R. A clinical and pathologic study of endometriosis of the lung. Surg Obstet Gynecol 1956;103:552-558.
  8. Rodman M.H., Jones C.W. Catamenial hemoptysis due to bronchial endometriosis. NEJM 1962;266:805.[Medline]
  9. Elloit D.L., Barker A.F., Dixon L. Catamenial hemoptysis. Chest 1985;87:687-688.[Abstract/Free Full Text]
  10. Ronnberg L., Ylostalo P. Treatment of pulmonary endometriosis with danazol. Acta Obstet Gynecol Scand 1981;60:77-78.[Medline]
  11. Rosenberg S.M., Riddick D.H. Successful treatment of catamenial hemoptysis with danazol. Obstet Gynecol 1981;57:130.[Medline]
  12. Suginami H., Hamada K., Yano K. A case of endometriosis of the lung treated with danazol. Obstet Gynecol 1985;66:68S.[Medline]



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