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92 Tüberküloz ve Toraks Dergisi 2008; 56(1): 92-95

Endobronchial metastases from a leiomyosarcoma of the uterus

Haldun Şükrü ERKAL1, Ayşen Sevgi ÖZTÜRK1, Lale KUTLUAY2, Dilek AYDIN3

1 Gazi Üniversitesi Tıp Fakültesi, Radyasyon Onkolojisi Anabilim Dalı,

2Bayındır Hastanesi, Patoloji Bölümü,

3 Bayındır Hastanesi, Göğüs Hastalıkları Bölümü, Ankara.

ÖZET

Uterus leiomiyosarkoması kaynaklı endobronşiyal metastaz

Altmışüç yaşında olan bir kadın hasta dispne, kuru öksürük ve göğüs ağrısı yakınmalarıyla başvurdu. Toraks bilgisayar- lı tomografisinde sol akciğerde tama yakın atelektazi bildirildi. Bronkoskopik incelemede, sol ana bronş mukozasında yer- leşen ve tama yakın tıkanıklığa yol açan bir kitle lezyonu saptandı. Hastanın tıbbi öyküsünde bir yıl önce uterus leiomi- yosarkoması için total abdominal histerektomi ve adjuvan radyoterapi ile tedavi edilmiş olduğu anlaşıldı. Hastaya metas- tatik leiomiyosarkoma tanısı konularak palyatif radyoterapi ile tedavi edildi. Hasta altı ay sonra toraksı ve pelvisi tutan yaygın hastalıkla başvurdu. Tedaviyi kabul etmeyen hasta bir ay sonra öldü. Uterus leiomiyosarkoması tanısı konulan hastalar, alışılmadık bölgelerde göreceli olarak erken metastazlar ile birlikte, saldırgan bir klinik gidiş izleyebilir.

Anahtar Kelimeler: Uterus, leiomiyosarkoma, endobronşiyal metastaz.

SUMMARY

Endobronchial metastases from a leiomyosarcoma of the uterus

Haldun Şükrü ERKAL1, Ayşen Sevgi ÖZTÜRK1, Lale KUTLUAY2, Dilek AYDIN3

1 Department of Radiation Oncology, Faculty of Medicine, Gazi University, Ankara, Turkey, 2 Department of Pathology, Bayındır Hospital, Ankara, Turkey,

3Department of Chest Diseases, Bayındır Hospital, Ankara, Turkey.

Yazışma Adresi (Address for Correspondence):

Dr. Haldun Şükrü ERKAL, İnönü Üniversitesi Tıp Fakültesi, Radyasyon Onkolojisi Anabilim Dalı, 44280, MALATYA - TURKEY

e-mail: hserkal@inonu.edu.tr

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Leiomyosarcomas are uncommon primary ute- rine neoplasms. Patients with leiomyosarcomas of the uterus might suffer subsequent recurren- ces in the form of distant metastasis at unusual sites following initial local treatment (1,2). We report the case of a woman presenting with an endobronchial metastases from a leiomyosarco- ma of the uterus that had received local treat- ment one year ago.

CASE REPORT

A 63-year-old woman presented with dyspnea, non-productive cough and chest pain. On physi- cal examination, she was found to have absent breath sounds over the left hemithorax. Chest X- ray revealed opacification of the left hemithorax, accompanied by left sided mediastinal shift and elevation of the left diaphragm. On computed tomography of the thorax, near total atelectasis of the left lung was reported (Figure 1). Her me- dical history revealed that she had received lo- cal treatment consisting of total abdominal hysterectomy followed by adjuvant radiation therapy for a leiomyosarcoma of the uterus one year ago. The patient was referred for bronchos- copic examination, which revealed near total obstruction of the left main bronchus at 2 cm from the carina by a mass lesion (Figure 2). Bi- opsies of the mass lesion were obtained during bronchoscopy. Histopathological sections of the biopsy specimen of the bronchial mucosa sho- wed a malignant mesenchymal tumor compo- sed of spindle cells with frequent mitotic figures, partial bundle formations and extensive necro- sis. Upon review of the specimen of the uterus together with the specimen of the bronchial mu- cosa, a histopathological diagnosis of “malig- nant mesenchymal tumor, consistent with le-

iomyosarcoma” was made (Figure 3). Endob- ronchial brachytherapy was not attempted ba- sed on the near total obstruction of the left ma- in bronchus on bronchoscopic examination. Ex- ternal beam radiation therapy with palliative in- tent was undertaken to the metastatic lesion as

Erkal HŞ, Öztürk AS, Kutluay L, Aydın D.

93 Tüberküloz ve Toraks Dergisi 2008; 56(1): 92-95 A 63-year-old woman presented with dyspnea, non-productive cough and chest pain. On computed tomography of the tho- rax, near total atelectasis of the left lung was reported. On bronchoscopic examination, a mass lesion was observed in the mucosa of the left main bronchus leading to near total obstruction. Her medical history revealed that she had been treated with total abdominal hysterectomy and adjuvant radiation therapy for a leiomyosarcoma of the uterus one year ago. She was diagnosed as having a metastatic leiomyosarcoma and was treated with palliative radiation therapy. Six months la- ter, she presented with disseminated disease involving the thorax and the pelvis. She refused further treatment and died one month later. Patients with leiomyosarcomas of the uterus might follow an aggressive clinical course, with relatively early metastases at unusual sites.

Key Words: Uterus, leiomyosarcoma, endobronchial metastases.

Figure 1. Near total atelectasis of the left lung was observed on computed tomography of the thorax (lung window).

Figure 2. Bronchoscopic examination revealed near total obstruction of the left main bronchus at 2 cm from the carina by a mass lesion.

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well as the mediastinum to a dose of 30 Gy de- livered in daily fractions of 3 Gy. The patient re- mained stable and asymptomatic for six months. At six months following the palliative radiation therapy course, she presented with dis- seminated disease involving the thorax, the pel- vis and the scalp. The patient refused further evaluation and treatment. She was discharged upon palliative medical measures and died at one month following her discharge.

DISCUSSION

Sarcomas comprise less than 5% of all primary neoplasms arising from the uterus. Of all sarco- mas arising from the uterus, 20 to 30% are le- iomyosarcomas that are commonly observed in women between 40 to 60 years of age, frequ- ently presenting with advanced local disease.

Leiomyosarcomas of the uterus can demonstra- te local spread by invasion and implantation ro- utes or metastatic spread by lymphatic and he- matogenous routes (1,2). As far as the metasta- tic spread by the hematogenous route is concer- ned, the lung has been reported to be the most frequently involved site in an autopsy study, li- kely in relation to being the first-pass organ for the venous drainage from the uterus. Metastatic spread to the lung has often been accompanied by metastatic spread to other sites, a finding that is consistent with the concept of cascade metas- tases from the first-pass organ (3).

Metastatic involvement of the lung by extrapul- monary malignant tumors is usually parenchy- mal, although endobronchial involvement can occasionally be observed. Endobronchial me- tastases from extrapulmonary malignant tumors are usually similar in appearance to primary bronchogenic malignant tumors. Fitzgerald has reviewed 17 patients with endobronchial metas- tases from various malignant tumors. One of these patients had been reported to have a le- iomyosarcoma arising from the forearm (4).

Only a few reports of patients with leiomyosar- comas of the uterus presenting with endobronc- hial metastases have been published. In 1978, Flynn and Kim have reported a patient with en- dobronchial metastases from a leiomyosarcoma of the uterus that had presented with severe dys- pnea from bronchial obstruction (5). Giudice et al., in 1979, have reported another patient with endobronchial metastases from a leiomyosarco- ma of the uterus that had presented with severe hemoptysis (6). In 1993, Gerst et al. have repor- ted a patient with a leiomyosarcoma of the ute- rus who had developed endobronchial metasta- ses and small bowel metastases eight years af- ter initial local treatment, namely total abdomi- nal hysterectomy (7).

The patient presented in this report developed metastases as early as one year after initial local treatment, at which time she was considered as having no evidence of local recurrence. The early development of metastases at multiple, and fairly unusual, sites in the absence of local recurrence is in contrary to the usual indolent clinical course depicted for leiomyosarcoma of the uterus, and is suggestive of an aggressive clinical course. The high mitotic rate observed in the histopathological examination of the mass lesion might prove a likely explanation for the predilection in the direction of the early develop- ment of metastases.

To come to the point, the patient presented in this report calls attention to the possibility that patients with leiomyosarcomas of the uterus might follow an aggressive clinical course and might develop relatively early metastases at unusual sites.

Endobronchial metastases from a leiomyosarcoma of the uterus

94 Tüberküloz ve Toraks Dergisi 2008; 56(1): 92-95

Figure 3. Histopathological sections of the bronchial mucosa showed a malignant mesenchymal tumor composed of spindle cells with frequent mitotic figu- res, partial bundle formations and extensive necrosis (HE, x100).

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Erkal HŞ, Öztürk AS, Kutluay L, Aydın D.

95 Tüberküloz ve Toraks Dergisi 2008; 56(1): 92-95 REFERENCES

1. Salazar OM, Bonfiglio TA, Patten SF, et al. Uterine sarco- mas: Analysis of failures with special emphasis on the use of adjuvant radiation therapy. Cancer 1978; 42:

1161-70.

2. Gadducci A, Landoni F, Sartori E, et al. Uterine leiomyo- sarcoma: Analysis of treatment failures and survival.

Gynecol Oncol 1996; 62: 25-32.

3. Rose PG, Piver MS, Tsukada Y, Lau T. Patterns of metas- tasis in uterine sarcoma: an autopsy study. Cancer 1989;

63: 935-8.

4. Fitzgerald RH. Endobronchial metastases. South Med J 1977; 70: 440-1.

5. Flynn KJ, Kim HS. Endobronchial metastasis of uterine leiomyosarcoma. J Am Med Assoc 1978; 240: 2080.

6. Giudice JC, Komansky H, Gordon R. Endobronchial me- tastasis of uterine leiomyosarcoma. J Am Med Assoc 1979; 241: 1684.

7. Gerst PH, Levy J, Swaminathan K, et al. Metastatic le- iomyosarcoma of the uterus: Unusual presentation of a case with late endobronchial and small bowel metasta- ses. Gynecol Oncol 1993; 49: 271-5.

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