- Email: [email protected]
Natural history and therapeutic implications of patients with catamenial hemoptysis
ARTICLE IN PRESS Respiratory Medicine (2007) 101, 1032–1036
Natural history and therapeutic implications of patients with catamenial hemoptysis$ Jeong-Seon Ryua,, Eun-Seop Songb, Kyung-Hee Leec, Jae Hwa Choa, Seung-Min Kwaka, Hong Lyeol Leea a
Department of Internal Medicine, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea b Obstetrics and Gynecology, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea c Diagnostic Radiology, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea Received 5 July 2006; accepted 21 August 2006 Available online 2 October 2006
KEYWORDS Hemoptysis; Endometriosis; Conservative treatment
Summary Evidence as to whether clinician has to give specific treatment in all patients of catamenial hemoptysis is unclear. We considered that the current treatment such as long-term usage of hormonal agent or surgery might be excessive for the catamenial hemoptysis. Therefore, we developed prospective observation study with observation strategy and follow-up for the patients. In sequential four patients of catamenial hemoptysis between December 2000 and November 2001, physical examination, chest X-ray, pelvic ultrasonogram and chest CT scan were taken at both the diagnosis and last follow-up. All patients were only observed without specific treatment within the limit of the possibility and followed for average 58 months. Mean age of patients was 23.5 years (range, 22–25 years). All patients have a history of undertaking one or two dilatations and curettages before diagnosis. The chest CT scans of all patients presented with ground-glass opacities of peripheral location that were disappeared without any residual lesion at last follow-up. Hemoptysis of two patients was spontaneously disappeared after 6 months. In the other two patients, it was greatly lessened in amount and frequency, then clinically insignificant in one. It was disappeared after subsequent 2 months and then relapsed two times in the late of follow-up of another patient.
$
This study was supported by INHA University Research Grant (INHA).
Corresponding author. Tel.: +82 32 890 3738; fax: +82 32 882 6578.
E-mail address: [email protected] (J.-S. Ryu). 0954-6111/$ - see front matter & 2006 Elsevier Ltd. All rights reserved. doi:10.1016/j.rmed.2006.08.021
ARTICLE IN PRESS Therapeutic implications of patients with catamenial hemoptysis
1033
This study suggests that observation only may be an alternative option in the treatment of catamenial hemoptysis. & 2006 Elsevier Ltd. All rights reserved.
Introduction Although endometriosis has been noted in 10–15% of women of reproductive age, catamenial hemoptysis is an extremely rare clinical subset. To our knowledge, only approximately 40 cases have been reported in the world. In the management of hemoptysis by other etiologies, surgical resection has been limitedly used in the massive, life-threatening hemoptysis. Most cases of catamenial hemoptysis in previous reports featured small quantities. There has been no report of a patient with catamenial hemoptysis showing massive or fatal hemoptysis.1 Furthermore, substantial rates of adverse effects and relapse have been reported in patients receiving hormonal agents in catameninal hemoptysis as well as pelvic endometriosis.2–5 Various modalities from long-term use of hormonal agents to surgical resection such as lobectomy have been tried in the treatment of catamenial hemoptysis.3,4,6–17 However, this information has largely arisen from case reports and experiences from pelvic endometriosis. Meanwhile, the natural history and pathogenesis of the catamenial hemoptysis remain largely unknown. We considered that the current treatment might be excessive for the catamenial hemoptysis and therefore developed observation strategy and follow-up for patients with catamenial hemoptysis. Here, we report our experience with four consecutive female patients with catamenial hemoptysis who did not receive any specific treatment for the hemoptysis. The follow-up results suggest that observation may be an alternative option in the treatment.
Materials and methods In this study, we initially included six patients who presented to the out-patient clinic of Inha university hospital between December 2000 and November 2001 and who was clinically suspicious of catamenial hemoptysis based upon the history. While three patients complained of recurrent bouts of hemoptysis during menstruation, the other three patients presented with her first episode of hemoptysis in the menstruation. During the follow-up, two of three patients were excluded because hemoptysis of two patients was not appeared in subsequent. In four patients, catamenial hemoptysis was clinically diagnosed with the characteristic clinical history and chest CT scan finding. Ground-glass opacities (GGO) that were observed on chest CT scan during menstruation were resolved during the subsequent intermenstrual period in all patients. In addition to physical examination, chest X-ray and laboratory testing, detailed history was taken by Song ES, a doctor of the department of obstetrics and gynecology. Pelvic ultrasonograms were undertaken at the time of diagnosis and last follow-up. To evaluate the residual change on the lung, chest CT scan was taken again at last follow-up. All four patients agreed with
our policy of observation only without specific treatment, hormonal agents or palliative surgical resection, for the hemopysis treatment. If hemoptysis became more severe in subsequent menstrual cycles, hormonal agents or surgical resection could be considered in a revised treatment plan. The Institutional Review Board of Inha university hospital approved the study and all patients provided informed consent (Fig. 1).
Results All four patients were single women (mean age, 23.5 years; range, 22–25 years) without history of pulmonary, cardiovascular or connective tissue diseases. No patients had prior history of hemoptysis in inter-menstrual period or endometriosis. Presenting symptoms other than hemoptysis were cough ing (patient nos. 1–3) and chest discomfort (patient no. 4). Physical examinations, chest X-ray, all laboratory tests, and pelvic ultrasonograms at both of diagnosis and last follow-up were normal in all four patients. One or two dilatations and curettages were undertaken before the diagnosis in all patients (Table 1). Sputum gram stain and culture for bacteria were negative on the sputum of three patients who could expectorate. Direct sputum examination produced negative results for acid-fast organisms and fungi. No endometrial cells were noted on the sputum. The mean duration of the concurrent occurrence of hemoptysis with menstruation prior to the diagnosis was 7.25 months (range, 0–15 months). Chest CT findings of all patients were GGO. No patients presented with consolidation, nodule(s) or findings suggesting other causes of hemoptysis such as tuberculosis, bronchiectasis, or pulmonary embolism on the CT scans. The mean duration of follow-up was 58 months (range, 54–66 months). After the diagnosis, patient no. 1, who presented with 5 mL/day of hemoptysis, subsequently experienced continuation of hemoptysis of streaky blood at approximate 3-month intervals, during the 55 months of follow-up. In October 2004, she complained of right lower abdominal pain and was diagnosed with pelvic endometriosis. Her pain was relieved with oral contraceptive for 4 months. Hemoptysis of patient no. 2, who presented with 5 mL/day of hemoptysis, also was greatly lessened in amount, then spontaneously disappeared 6 months later, and did not recur during the 54 months of follow-up. She received second dilatation and curettage in August 2003 and did not take any hormonal agents during the follow-up. Patient no. 3 presented with two episodes of hemoptysis (30 and 15 mL/day, respectively). After two more subsequent episodes of hemoptysis during menstruation, it spontaneously disappeared and then recurred two times in August 2005 (5 mL/day) and January 2006 (5 mL/day) during 57 months of follow-up. She also did not take any hormonal agents during the follow-up. In patient no. 4, who presented with the first episode of hemoptysis (10 mL/day), hemopty-
ARTICLE IN PRESS 1034
J.-S. Ryu et al.
Figure 1 Chest CT scans at the diagnosis, subsequent inter-menses period and last follow-up in all four patients of catamenial hemoptysis in which the ground-glass opacities were disappeared without any residual lesion.
sis spontaneously disappeared after six subsequent episodes during menstruation. It did not recur during the 66 months of follow-up. She got married in October 2004 and gave birth in July 2005.
Discussion This study demonstrated that observation only could be an alterative option in the treatment of catamenial hemoptysis. Hemoptysis is a very commonly encountered problem for most clinicians. The decision on the treatment of hemoptysis by other etiologies has been made with the information of the natural history of the causing diseases and its degree of severity as measured by the amount. In contrast with the observation only strategy presented in this study, hormonal agents or surgical resection were given to most reported patients of catameninal hemoptysis.3,4,6–17 Hormonal agents, including danazol, gonadotropin-releasing hormone agonists, and oral contraceptives, have been considered as the first option by clinicians.3,4,6,10–12,16–18 In patients showing adverse effects from the hormonal agent or recurrence, surgical resection has been used as the second option.3,4,7 In an early report lobectomy was tried in the first treatment for hemoptysis. The recent trend has been toward palliative treatment. Parenchymal preserving surgical procedures such as videoassisted thoracoscopic surgery were used to treat the patients having single peripheral lesion.13,14 In addition, palliative local treatment using Nd-YAG laser was reported
in the patients of centrally located, tracheobronchial endometriosis.19,20 No data has been reported on the percentage of patients who suffer from adverse reactions, or show recurrence after hormonal agents or surgery. Unfortunately, current knowledge on treatment of catamenial hemoptysis has arisen only from the limited number of reported cases or experiences of pelvic endometriosis, rather than from well designed, controlled study. Because most patients from case series already reported were almost in the age of hoping to be pregnant, hormonal agents could be hard to be the treatment option in the patients. Determination of whether specific treatment is required for all patients of catamenial hemoptysis remains unclear for the following reasons. Firstly, there has been no report of massive hemoptysis or mortality from catamenial hemoptysis. Secondly, it is unknown whether the implanted endometrial cell behaves like normal tissue and operates under hormonal influence or whether it is spontaneously shed. Thirdly, the experiences from patients of pelvic endometriosis showed that laparoscopic surgical resection as a definitive treatment was also associated with substantial rates of recurrence, and that the use of hormonal agents also carries adverse effects.5,21 In an average 58 months of follow-up, hemoptysis spontaneously disappeared after subsequent continuations of 2 or 6 months in three patients (patients nos. 2–4) although one patient (no 3.) recurred two times synchronously with her menstruation in the late of follow-up. Two of three patients (patients nos. 2 and 4) became pregnant in the late of follow-up. The fact that pregnancy is a kind of
ARTICLE IN PRESS
D&C ¼ dilatation and curettage; OC ¼ oral contraceptive; NE ¼ not evaluable; RUL ¼ right upper lobe, RLL ¼ right lower lobe, RML ¼ right middle lobe.
Yes, pregnancy 42 No 0 10 and 35 No 4/December 2000
122/12
Never
2/0
46 No NE 2 9 and 21 No Never
2/0
Yes
2/November 24/13 2001 3/August 2001 25/13
Current
1/0
23
15
No
Yes, D&C
16
Post. Seg. of RUL Anterobasal Seg. of RLL Apical Seg. of RUL Post. Seg. of RUL Apical Seg. of RUL; Medial Seg. of RML; Sup. Seg. of RLL No Yes, OC No 12 20 Yes 23/13 1/October 2001
Ex
1/0
Duration prior to diagnosis (months) D&C before the diagnosis (months) Gravidity/ parity Dysmenorrhea Smoking history Age (years)/ age of menarche (years) Patient no./ date of diagnosis
Table 1
Clinical characteristics of catamenial hemoptysis at the diagnosis in four patients.
Endometrial Hormone cells in sputum treatment
Location on Remission before hormonal chest CT scan treatment (months)
Therapeutic implications of patients with catamenial hemoptysis
1035 hormonal treatment of catamenial hemoptysis could be confounding factor in drawing a conclusion from this study. Even if so, the hemoptysis was however ceased for 16 and 42 months without specific treatment before the pregnancy, respectively. The chest CT scans in subsequent inter-menstrual periods and at last follow-up were clear without any residual change in all patients. These findings may suggest the feasibility of spontaneous resolution of implanted endometrial cells in lung parenchyma, in contrast to the infrequent event in pelvic endometriosis mainly induced by tubal regurgitation. In the present study, all patients showed a relatively small extent of GGO peripherally located on the chest CT scan and underwent one or two episodes of dilatation and curettage before onset of hemoptysis. Sampson’s theory of transdiaphragmatic spread through tubal regurgitation seems to be a lower possibility as pathogenic mechanism of all four patients.22 Tubal regurgitation is a frequent event observed in 76–90% of women.23,24 According to this theory, the lower lobe of the lung would be expected to be more likely affected and a larger amount of endometrial cells would be expected to have migrated to the lung rather than in lymphovascular embolization. However, the present report of this was limited in making any definition of the pathogenic mechanism. Therefore, a basic investigation is needed to elucidate the mechanism and the behavior of implanted endometrial cells. Although diagnostic guideline of catamenial hemoptysis is not available, present study has a limitation of not performing flexible bronchoscopy that would be helpful in identifying the source of bleeding, excluding other tracheobronchial lesions, trying to obtain a pathological confirmation or the possibility of centrally located airway endometriosis. This is first study of catamenial hemoptysis treatement restricted to observation only with approximately five years of follow-up duration and looking at the natural history with a limitation of small number of cases. If a patient has massive hemoptysis from catamenial hemoptysis, the benign results of this study, in which no patient presented with massive hemoptysis, may not be applicable and that intervention would be appropriate. We reported that observation only could be an alternative to surgical resection or long-term usage of hormonal agents, especially in the patients of hoping to be pregnant, or unwilling to take surgical resection.
References 1. Alifano M, Trisolini R, Cancellieri A, et al. Thoracic endometriosis: current knowledge. Ann Thorac Surg 2006;81:761–9. 2. Olive DL, Schwartz LB. Endometriosis. N Engl J Med 1993;328:1759–69. 3. Suginami H, Hamada K, Yano K. A case of endometriosis of the lung treated with danazol. Obstet Gynecol 1985;66:68S–71S. 4. Johnson III WM, Tyndal CM. Pulmonary endometriosis: treatment with danazol. Obstet Gynecol 1987;69:506–7. 5. Buttram Jr VC, Belue JB, Reiter R. Interim report a study of danazol for the treatment of endometriosis. Fertil Steril 1982;37:478–83.
ARTICLE IN PRESS 1036 6. Elliot DL, Barker AF, Dixon LM. Catamenial hemoptysis. New methods of diagnosis and therapy. Chest 1985; 87:687–8. 7. Kristianen K, Fjeld NB. Pulmonary endometriosis causing haemoptysis. Report of a case treated with lobectomy. Scand J Thorac Cardiovasc Surg 1993;27:113–5. 8. Joseph J, Sahn SA. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996;100:164–70. 9. Terada Y, Chen F, Shoji T, et al. A case of endobronchial endometriosis treated by subsegmentectomy. Chest 1999;115:1475–8. 10. Yu Z, Fleischman JK, Rahman HM, et al. Catamenial hemoptysis and pulmonary endometriosis: a case report. Mt Sinai J Med 2002;69:261–3. 11. Katoh O, Yamada H, Aoki Y, et al. Utility of angiograms in patients with catamenial hemoptysis. Chest 1990; 98:1296–7. 12. Hope-Gill B, Prathibha BV. Catamenial haemoptysis and clomiphene citrate therapy. Thorax 2003;58:89–90. 13. Cassina PC, Hauser M, Kacl G, et al. Catamenial hemoptysis. Diagnosis with MRI. Chest 1997;111:1447–50. 14. Park YB, Heo GM, Moon HK, et al. Pulmonary endometriosis resected by video-assisted thoracoscopic surgery. Respirology 2006;11:221–3. 15. Ronnberg L, Ylostalo P. Treatment of pulmonary endometriosis with danazol. Acta Obstet Gynecol Scand 1981;60:77–8.
J.-S. Ryu et al. 16. Espaulella J, Armengol J, Bella F, et al. Pulmonary endometriosis: conservative treatment with GnRH agonists. Obstet Gynecol 1991;78:535–7. 17. Wang HC, Kuo PH, Kuo SH, et al. Catamenial hemoptysis from tracheobronchial endometriosis: reappraisal of diagnostic value of bronchoscopy and bronchial brush cytology. Chest 2000;118:1205–8. 18. Rosenberg SM, Riddick DH. Successful treatment of catamenial hemoptysis with danazol. Obstet Gynecol 1981;57:130–2. 19. Puma F, Carloni A, Casucci G, et al. Successful endoscopic NdYAG laser treatment of endobronchial endometriosis. Chest 2003;124:1168–70. 20. Ozvaran MK, Baran R, Sogukpmar O, et al. Histopathological diagnosis of endobronchial endometriosis treated with argon laser. Respirology 2006;11:348–50. 21. Milingos S, Protopapas A, Drakakis P, et al. Laparoscopic management of patients with endometriosis and chronic pelvic pain. Ann N Y Acad Sci 2003;997:269–73. 22. Sampson J. Peritoneal endometriosis due to the menstrual dissemination of endometrial tissue into the peritoneal cavity. Am J Obstet Gynecol 1927;14:422–72. 23. Blumenkrantz MJ, Gallagher N, Bashore RA, et al. Retrograde menstruation in women undergoing chronic peritoneal dialysis. Obstet Gynecol 1981;57:667–70. 24. Halme J, Hammond MG, Hulka JF, et al. Retrograde menstruation in healthy women and in patients with endometriosis. Obstet Gynecol 1984;64:151–4.
Natural history and therapeutic implications of patients with catamenial hemoptysis$ Jeong-Seon Ryua,, Eun-Seop Songb, Kyung-Hee Leec, Jae Hwa Choa, Seung-Min Kwaka, Hong Lyeol Leea a
Department of Internal Medicine, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea b Obstetrics and Gynecology, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea c Diagnostic Radiology, College of Medicine, Inha University, Hospital 7-206, 3-Ga, Shinheung Dong, Jung Gu, Inchon 400-103, Republic of Korea Received 5 July 2006; accepted 21 August 2006 Available online 2 October 2006
KEYWORDS Hemoptysis; Endometriosis; Conservative treatment
Summary Evidence as to whether clinician has to give specific treatment in all patients of catamenial hemoptysis is unclear. We considered that the current treatment such as long-term usage of hormonal agent or surgery might be excessive for the catamenial hemoptysis. Therefore, we developed prospective observation study with observation strategy and follow-up for the patients. In sequential four patients of catamenial hemoptysis between December 2000 and November 2001, physical examination, chest X-ray, pelvic ultrasonogram and chest CT scan were taken at both the diagnosis and last follow-up. All patients were only observed without specific treatment within the limit of the possibility and followed for average 58 months. Mean age of patients was 23.5 years (range, 22–25 years). All patients have a history of undertaking one or two dilatations and curettages before diagnosis. The chest CT scans of all patients presented with ground-glass opacities of peripheral location that were disappeared without any residual lesion at last follow-up. Hemoptysis of two patients was spontaneously disappeared after 6 months. In the other two patients, it was greatly lessened in amount and frequency, then clinically insignificant in one. It was disappeared after subsequent 2 months and then relapsed two times in the late of follow-up of another patient.
$
This study was supported by INHA University Research Grant (INHA).
Corresponding author. Tel.: +82 32 890 3738; fax: +82 32 882 6578.
E-mail address: [email protected] (J.-S. Ryu). 0954-6111/$ - see front matter & 2006 Elsevier Ltd. All rights reserved. doi:10.1016/j.rmed.2006.08.021
ARTICLE IN PRESS Therapeutic implications of patients with catamenial hemoptysis
1033
This study suggests that observation only may be an alternative option in the treatment of catamenial hemoptysis. & 2006 Elsevier Ltd. All rights reserved.
Introduction Although endometriosis has been noted in 10–15% of women of reproductive age, catamenial hemoptysis is an extremely rare clinical subset. To our knowledge, only approximately 40 cases have been reported in the world. In the management of hemoptysis by other etiologies, surgical resection has been limitedly used in the massive, life-threatening hemoptysis. Most cases of catamenial hemoptysis in previous reports featured small quantities. There has been no report of a patient with catamenial hemoptysis showing massive or fatal hemoptysis.1 Furthermore, substantial rates of adverse effects and relapse have been reported in patients receiving hormonal agents in catameninal hemoptysis as well as pelvic endometriosis.2–5 Various modalities from long-term use of hormonal agents to surgical resection such as lobectomy have been tried in the treatment of catamenial hemoptysis.3,4,6–17 However, this information has largely arisen from case reports and experiences from pelvic endometriosis. Meanwhile, the natural history and pathogenesis of the catamenial hemoptysis remain largely unknown. We considered that the current treatment might be excessive for the catamenial hemoptysis and therefore developed observation strategy and follow-up for patients with catamenial hemoptysis. Here, we report our experience with four consecutive female patients with catamenial hemoptysis who did not receive any specific treatment for the hemoptysis. The follow-up results suggest that observation may be an alternative option in the treatment.
Materials and methods In this study, we initially included six patients who presented to the out-patient clinic of Inha university hospital between December 2000 and November 2001 and who was clinically suspicious of catamenial hemoptysis based upon the history. While three patients complained of recurrent bouts of hemoptysis during menstruation, the other three patients presented with her first episode of hemoptysis in the menstruation. During the follow-up, two of three patients were excluded because hemoptysis of two patients was not appeared in subsequent. In four patients, catamenial hemoptysis was clinically diagnosed with the characteristic clinical history and chest CT scan finding. Ground-glass opacities (GGO) that were observed on chest CT scan during menstruation were resolved during the subsequent intermenstrual period in all patients. In addition to physical examination, chest X-ray and laboratory testing, detailed history was taken by Song ES, a doctor of the department of obstetrics and gynecology. Pelvic ultrasonograms were undertaken at the time of diagnosis and last follow-up. To evaluate the residual change on the lung, chest CT scan was taken again at last follow-up. All four patients agreed with
our policy of observation only without specific treatment, hormonal agents or palliative surgical resection, for the hemopysis treatment. If hemoptysis became more severe in subsequent menstrual cycles, hormonal agents or surgical resection could be considered in a revised treatment plan. The Institutional Review Board of Inha university hospital approved the study and all patients provided informed consent (Fig. 1).
Results All four patients were single women (mean age, 23.5 years; range, 22–25 years) without history of pulmonary, cardiovascular or connective tissue diseases. No patients had prior history of hemoptysis in inter-menstrual period or endometriosis. Presenting symptoms other than hemoptysis were cough ing (patient nos. 1–3) and chest discomfort (patient no. 4). Physical examinations, chest X-ray, all laboratory tests, and pelvic ultrasonograms at both of diagnosis and last follow-up were normal in all four patients. One or two dilatations and curettages were undertaken before the diagnosis in all patients (Table 1). Sputum gram stain and culture for bacteria were negative on the sputum of three patients who could expectorate. Direct sputum examination produced negative results for acid-fast organisms and fungi. No endometrial cells were noted on the sputum. The mean duration of the concurrent occurrence of hemoptysis with menstruation prior to the diagnosis was 7.25 months (range, 0–15 months). Chest CT findings of all patients were GGO. No patients presented with consolidation, nodule(s) or findings suggesting other causes of hemoptysis such as tuberculosis, bronchiectasis, or pulmonary embolism on the CT scans. The mean duration of follow-up was 58 months (range, 54–66 months). After the diagnosis, patient no. 1, who presented with 5 mL/day of hemoptysis, subsequently experienced continuation of hemoptysis of streaky blood at approximate 3-month intervals, during the 55 months of follow-up. In October 2004, she complained of right lower abdominal pain and was diagnosed with pelvic endometriosis. Her pain was relieved with oral contraceptive for 4 months. Hemoptysis of patient no. 2, who presented with 5 mL/day of hemoptysis, also was greatly lessened in amount, then spontaneously disappeared 6 months later, and did not recur during the 54 months of follow-up. She received second dilatation and curettage in August 2003 and did not take any hormonal agents during the follow-up. Patient no. 3 presented with two episodes of hemoptysis (30 and 15 mL/day, respectively). After two more subsequent episodes of hemoptysis during menstruation, it spontaneously disappeared and then recurred two times in August 2005 (5 mL/day) and January 2006 (5 mL/day) during 57 months of follow-up. She also did not take any hormonal agents during the follow-up. In patient no. 4, who presented with the first episode of hemoptysis (10 mL/day), hemopty-
ARTICLE IN PRESS 1034
J.-S. Ryu et al.
Figure 1 Chest CT scans at the diagnosis, subsequent inter-menses period and last follow-up in all four patients of catamenial hemoptysis in which the ground-glass opacities were disappeared without any residual lesion.
sis spontaneously disappeared after six subsequent episodes during menstruation. It did not recur during the 66 months of follow-up. She got married in October 2004 and gave birth in July 2005.
Discussion This study demonstrated that observation only could be an alterative option in the treatment of catamenial hemoptysis. Hemoptysis is a very commonly encountered problem for most clinicians. The decision on the treatment of hemoptysis by other etiologies has been made with the information of the natural history of the causing diseases and its degree of severity as measured by the amount. In contrast with the observation only strategy presented in this study, hormonal agents or surgical resection were given to most reported patients of catameninal hemoptysis.3,4,6–17 Hormonal agents, including danazol, gonadotropin-releasing hormone agonists, and oral contraceptives, have been considered as the first option by clinicians.3,4,6,10–12,16–18 In patients showing adverse effects from the hormonal agent or recurrence, surgical resection has been used as the second option.3,4,7 In an early report lobectomy was tried in the first treatment for hemoptysis. The recent trend has been toward palliative treatment. Parenchymal preserving surgical procedures such as videoassisted thoracoscopic surgery were used to treat the patients having single peripheral lesion.13,14 In addition, palliative local treatment using Nd-YAG laser was reported
in the patients of centrally located, tracheobronchial endometriosis.19,20 No data has been reported on the percentage of patients who suffer from adverse reactions, or show recurrence after hormonal agents or surgery. Unfortunately, current knowledge on treatment of catamenial hemoptysis has arisen only from the limited number of reported cases or experiences of pelvic endometriosis, rather than from well designed, controlled study. Because most patients from case series already reported were almost in the age of hoping to be pregnant, hormonal agents could be hard to be the treatment option in the patients. Determination of whether specific treatment is required for all patients of catamenial hemoptysis remains unclear for the following reasons. Firstly, there has been no report of massive hemoptysis or mortality from catamenial hemoptysis. Secondly, it is unknown whether the implanted endometrial cell behaves like normal tissue and operates under hormonal influence or whether it is spontaneously shed. Thirdly, the experiences from patients of pelvic endometriosis showed that laparoscopic surgical resection as a definitive treatment was also associated with substantial rates of recurrence, and that the use of hormonal agents also carries adverse effects.5,21 In an average 58 months of follow-up, hemoptysis spontaneously disappeared after subsequent continuations of 2 or 6 months in three patients (patients nos. 2–4) although one patient (no 3.) recurred two times synchronously with her menstruation in the late of follow-up. Two of three patients (patients nos. 2 and 4) became pregnant in the late of follow-up. The fact that pregnancy is a kind of
ARTICLE IN PRESS
D&C ¼ dilatation and curettage; OC ¼ oral contraceptive; NE ¼ not evaluable; RUL ¼ right upper lobe, RLL ¼ right lower lobe, RML ¼ right middle lobe.
Yes, pregnancy 42 No 0 10 and 35 No 4/December 2000
122/12
Never
2/0
46 No NE 2 9 and 21 No Never
2/0
Yes
2/November 24/13 2001 3/August 2001 25/13
Current
1/0
23
15
No
Yes, D&C
16
Post. Seg. of RUL Anterobasal Seg. of RLL Apical Seg. of RUL Post. Seg. of RUL Apical Seg. of RUL; Medial Seg. of RML; Sup. Seg. of RLL No Yes, OC No 12 20 Yes 23/13 1/October 2001
Ex
1/0
Duration prior to diagnosis (months) D&C before the diagnosis (months) Gravidity/ parity Dysmenorrhea Smoking history Age (years)/ age of menarche (years) Patient no./ date of diagnosis
Table 1
Clinical characteristics of catamenial hemoptysis at the diagnosis in four patients.
Endometrial Hormone cells in sputum treatment
Location on Remission before hormonal chest CT scan treatment (months)
Therapeutic implications of patients with catamenial hemoptysis
1035 hormonal treatment of catamenial hemoptysis could be confounding factor in drawing a conclusion from this study. Even if so, the hemoptysis was however ceased for 16 and 42 months without specific treatment before the pregnancy, respectively. The chest CT scans in subsequent inter-menstrual periods and at last follow-up were clear without any residual change in all patients. These findings may suggest the feasibility of spontaneous resolution of implanted endometrial cells in lung parenchyma, in contrast to the infrequent event in pelvic endometriosis mainly induced by tubal regurgitation. In the present study, all patients showed a relatively small extent of GGO peripherally located on the chest CT scan and underwent one or two episodes of dilatation and curettage before onset of hemoptysis. Sampson’s theory of transdiaphragmatic spread through tubal regurgitation seems to be a lower possibility as pathogenic mechanism of all four patients.22 Tubal regurgitation is a frequent event observed in 76–90% of women.23,24 According to this theory, the lower lobe of the lung would be expected to be more likely affected and a larger amount of endometrial cells would be expected to have migrated to the lung rather than in lymphovascular embolization. However, the present report of this was limited in making any definition of the pathogenic mechanism. Therefore, a basic investigation is needed to elucidate the mechanism and the behavior of implanted endometrial cells. Although diagnostic guideline of catamenial hemoptysis is not available, present study has a limitation of not performing flexible bronchoscopy that would be helpful in identifying the source of bleeding, excluding other tracheobronchial lesions, trying to obtain a pathological confirmation or the possibility of centrally located airway endometriosis. This is first study of catamenial hemoptysis treatement restricted to observation only with approximately five years of follow-up duration and looking at the natural history with a limitation of small number of cases. If a patient has massive hemoptysis from catamenial hemoptysis, the benign results of this study, in which no patient presented with massive hemoptysis, may not be applicable and that intervention would be appropriate. We reported that observation only could be an alternative to surgical resection or long-term usage of hormonal agents, especially in the patients of hoping to be pregnant, or unwilling to take surgical resection.
References 1. Alifano M, Trisolini R, Cancellieri A, et al. Thoracic endometriosis: current knowledge. Ann Thorac Surg 2006;81:761–9. 2. Olive DL, Schwartz LB. Endometriosis. N Engl J Med 1993;328:1759–69. 3. Suginami H, Hamada K, Yano K. A case of endometriosis of the lung treated with danazol. Obstet Gynecol 1985;66:68S–71S. 4. Johnson III WM, Tyndal CM. Pulmonary endometriosis: treatment with danazol. Obstet Gynecol 1987;69:506–7. 5. Buttram Jr VC, Belue JB, Reiter R. Interim report a study of danazol for the treatment of endometriosis. Fertil Steril 1982;37:478–83.
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