Japanese Journal of Clinical Oncology 33:198-201 (2003)
© 2003 Foundation for Promotion of Cancer Research
Iridium-192 Brachytherapy for Hemorrhagic Angiosarcoma of the Scalp: a Case Report
Departments of 1 Radiology, 2 Dermatology and 3 Dental Radiology, Iwate Medical University, Morioka, Japan
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ABSTRACT |
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A 60-year-old woman presented with multicentric skin tumors of the head. The histologically proven hemangioendothelioma was bleeding as a result of disseminated coagulopathy. In addition to immunotherapy, 6 MeV electron beam radiotherapy was used for the purpose of hemostasis with a single portal, 20 x 20 cm in size, covering the whole scalp from the top of the head. The radiotherapy was discontinued after 39 Gy/13 fractions/20 days because of the progress in size of a peripheral tumor and the stability of coagulopathy. After another electron boost delivery by two portals focused to exophytic parietal and temporal tumors of 20 Gy/10 fractions, high dose rate brachytherapy with a surface mold technique was performed, 3 Gy/fraction, four fractions/week, for a total of 36 Gy. It induced partial regression of the tumor and complete recovery of platelet counts from 2.5 x 104 to 18.2 x 104. The tumor disappeared in 3 months. No late side effects occurred, except for permanent alopecia. The patient developed a cervical lymph node metastasis 1 year after and marginal recurrence 2 years after the initial treatment. Both recurrent tumors were successfully treated by 4 MV external photons of 60 Gy/20 fractions/46 days and electron beam irradiation of 60 Gy/20 fractions/29 days, respectively. She has remained disease free for 3 years after the initial presentation.
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INTRODUCTION |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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Angiosarcoma of the scalp is a highly aggressive tumor in aged populations. When it is large, primary treatment is radiotherapy, but the prognosis is usually dismal (1). Owing to the extension of the tumor beyond the areas treated, marginal recurrence is frequent (2). Various methods have been developed to treat homogeneously an extensive superficial curved volume, such as electron beam irradiation by multiple fields (3,4) or rotational portal (5), a combination of electron and photon beam techniques (6) and photon arc treatments (7). We adopted a high dose rate iridium-192 brachytherapy with a surface mold technique (8) after a failure in local control of an extended angiosarcoma of the skull by electron beam irradiation with a single portal.
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CASE REPORT |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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A 60-year-old woman presented to the dermatologist with multicentric, purple–red nodules spreading widely over the parietal scalp. A bleeding ulcer was found in the center of the conglomerated nodule. A bulky skip lesion formed an exophytic mass, 5 cm in diameter, located in the left temporal region. The patient recalled hitting her head on an iron pole 3 months before the visit, resulting in a head injury. Histological examination of an excision biopsy specimen made the diagnosis of malignant hemangioendothelioma. No lymph node or distant organ metastasis was found in image studies including chest radiography, ultrasonography of the neck, gallium scintigram and computed tomography (CT) of the head and neck. Platelet counts were less than 30 000/mm3. Blood coagulation tests showed prolongation of prothrombin time (PT), partial prothrombin time (PTT) and increased fibrin/fibrinogen degeneration products (FDP) and D-dimer. Frequent blood platelet transfusion resulted in only a small temporary recovery.
As immunotherapy by systemic injection of interleukin 2 failed to improve coagulopathy, radiotherapy was added using a 6 MeV electron beam by a single field, 20 x 20 cm in size, covering the entire scalp skin from the top of the head. The prescription dose per fraction was 3 Gy, four fractions per week. It was discontinued when a total of 39 Gy had been delivered over 20 days because the left temporal tumor progressed in size and coagulopathy still remained unchanged. The isodose distribution calculated by a three-dimensional treatment-planning device (Cadplan version 3.3, Varian) revealed a significant underdosage in the growing tumor (Fig. 1). We then decided to adopt the iridium-192 surface mold technique to avoid the underdosage. To make the surface mold close to the entire tumor, the thickness of the lesion should be as even as possible. We then delivered further electron beam irradiation on the parietal and left temporal areas separately with each portal(s), 10 cm x 10 cm in size, at 2 Gy/fraction, five fractions/week, a total of 20 Gy over 13 days. Although the left temporal tumor ceased to grow, the prepared therapy flattened the targeted tumor far less than expected. We made a helmet with a heat-moldable plastic facemask instead of the plaster casting tape described originally (8). The left temporal part of the mask was cut to prevent its separation from the skin surface due to the exophytic tumor. Fourteen flexible polyethylene tubes of 5 Fr. gauge were fixed in parallel along the sagittal plane on the inner surface of the mask. For delivery on the left temporal tumor exposed beyond the mask, four other guide tubes were separately arranged on the mask (Fig. 2). Treatment planning was performed on a device (PLATO-BPS version 13.1, Nucletron) using the CT images obtained as the mold put on her head. Active source dwell positions were determined to cover the entire tumor at 5 mm intervals in each applicator. The reference points were set at 20 mm beneath the surface of the tumor. For the left temporal tumor, the depths were increased to 30 mm. Geometric optimization in volume was calculated to obtain a uniform dose distribution (Fig. 3). From the next day to the end of electron radiotherapy, a total of 12 fractions, three fractions per week, a total of 36 Gy was delivered over 33 days. The total dose used in brachytherapy was limited because it had already reached the threshold dose for skin at the end of the electron beam radiotherapy and the brain was also considerably irradiated in this method. The tumor showed an apparent decrease in volume during the treatment, especially in the left temporal portion, which became almost flat.
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The patient complained of pain in the irradiated area of the head and needed analgesics for several months after the completion of the treatment. About 2 weeks after the end of therapy, the blood platelet count began to increase and it reached a normal value in 2 months. At the same time, FDP and D-dimer decreased. The tumor had almost disappeared 3 months after the brachytherapy (Fig. 4). Alopecia occurred and was irreversible in the irradiated area.
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One year later, a metastatic nodule, 4 x 3 cm in size, developed at the lower pole of the parotid gland. It was treated by 4 MV photon irradiation with anterior–posterior opposed pair portals of 3 Gy/fraction, four fractions/week, a total dose of 60 Gy over 46 days. The tumor showed almost complete regression at the end of therapy.
Two years later, several dusky, purple–red plaques appeared on the skin of the right temporal area, bordering the bold parietal skin previously irradiated; 4 MV electron beam irradiation of 3 Gy/fraction, four fractions/week and a total dose of 60 Gy over 29 days, was delivered by a 15 x 15 cm single portal with 5 mm bolus. At the end of this treatment the plaques became flat and they disappeared in 3 months. The patient is alive and free from disease now, 6 months after the last radiotherapy and 3 years after initial treatment. Recent plain radiography and MRI of the head did not indicate any damage to the skull or brain.
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DISCUSSION |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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The prognosis of angiosarcoma of the head and neck is generally poor. Tumor size at presentation is a significant indicator of prognosis in angiosarcoma of the scalp. In a multi-institutional study of 72 patients, the 5-year survival rate of angiosarcoma of the head and neck was 12%, with a median survival time of 15 months. Among them, the 2-year survival rate of patients with lesions greater than 10 cm was only 4% (2). In other large series, all patients with unresectable tumors died of the disease in a median time of 18 months after diagnosis (1). Although our case belongs to those with a poor prognosis, she still survives more than 3 years with controlled primary and loco-regional recurrent tumor.
Another significant feature of this case is intractable thrombocytopenia noted at presentation. The elevated levels of serum FDP and D-dimer confirmed that it resulted from disseminated intravascular coagulopathy (DIC). Since it was resolved at the regression of the tumor, our case was associated with Kasabach–Merritt phenomenon, which is not well reported in angiosarcoma (2).
One year after the complete remission of the primary scalp lesion, cervical lymph node metastasis occurred. The rate of regional nodal involvement, reported to occur in 20–30% of cases, is higher than that of the most sarcomas (9). Some authors reported parotid and neck nodal bed irradiation with electrons (10). Since subclinical disease can be controlled by moderate doses of radiation (10), prophylactic neck irradiation might have been indicated in such cases with large angiosarcoma.
Two years after the primary remission, marginal recurrence occurred in the right temporal skin where it had not been irradiated enough. The tumors usually extend transdermally to far wider distances than their clinical appearance (11), and treatment with a wide margin, such as total scalp irradiation, is rightly recommended.
In this wide scalp lesion, the therapeutic goal is not easily achieved, mainly owing to the geometry of the head and the close proximity of the scalp to the brain. In electron beam radiotherapy for this disease, various methods have been proposed using multiple abutting fields or arc therapy. Technical mismatch of the initial electron beam treatment was apparent in this case. The skin surface covered by single electron beam fields must be kept from being too far angled away from perpendicular incidence. In addition, lack of bolus or scattering filter might cause significant underdosage in the surface area of the tumor.
The role of radiotherapy as a primary radical treatment of this rare tumor is poorly documented and the optimum dose is unknown. In a large series of retrospective studies, the doses ranged from 44 Gy/11 fractions/17 days to 61 Gy/16 fractions/45 days (1). In another, a dose to the tumor bed 60 Gy in 2 Gy fractions was recommended (2). In this case, the sum of the dose delivered to the tumor reached a maximum of 95 Gy in the initial session.
The contiguous delivery of sufficient doses in this mold brachytherapy should avoid recurrences due to underdosage at the match-lines in electron beam radiotherapy using multiple fields (2). When compared with modern tomotherapy (12) requiring labor-intensive and complex techniques, uniformity in dose delivery may be equivalent (13). In addition, by shifting the reference dose point according to the height of the tumor, we can adjust the minimum required dose line along the base of the tumor. The manipulation resulted in significantly higher dose delivery in the left temporal tumor, which showed a faster regression than the others. The very steep dose gradient in brachytherapy is another advantage over electron beam radiotherapy, which is overlooked when it is evaluated only by the prescription of dosage.
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FOOTNOTES |
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+ For reprints and all correspondence: Ryuji Nakamura, Department of Radiology, Iwate Medical University, 19-1 Uchimaru, Morioka, Iwate, 020-8505 Japan. E-mail: rnakamur{at}iwate-med.ac.jp
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REFERENCES |
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Received August 28, 2002; accepted February 7, 2003
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