Ann Thorac Surg 2003;75:1302-1304
© 2003 The Society of Thoracic Surgeons
Case report
Massive hemoptysis caused by tracheal hemangioma treated with interventional radiology
Antonio Ríos Zambudio, MDa*,
Maria Jose Roca Calvo, PhDa,
Juan Torres Lanzas, PhDa,
J. García Medina, MDb,
Pascual Parrilla Paricio, PhDa
a Department of Surgery, Thoracic Surgery Unit, Murcia, Spain
b Interventional Radiology Unit, Hospital Universitario Virgen de la Arrixaca, Murcia, Spain
Accepted for publication October 14, 2002.
* Address reprint requests to Dr Zambudio, Avenida de la Libertad No. 208, Casillas Murcia 30007, Spain
e-mail: arzrios{at}teleline.es
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Abstract
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Capillary hemangiomas of the tracheobronchial tree are extremely rare in adults, with hemoptysis being one of the most serious forms of presentation. An operation has been the treatment of choice, although it does involve high rates of morbidity and mortality, especially in emergency situations such as massive hemoptysis, which has led to the search for other therapeutic alternatives. There is no experience with embolization by interventional radiology when the hemoptysis is tracheal in origin, caused partly because the infrequency of this pathology; however, the foundations for it have been laid with the development of embolization for bronchopulmonary pathology. We report a case of a tracheal capillary hemangioma in a 66-year-old woman diagnosed with idiopathic thrombopenic purpura, which began as a massive hemoptysis and was treated successfully with embolization by interventional radiology. There has been no recurrence of the bleeding after 1 year’s follow-up, and the patient’s control fibrobronchoscopy is normal.
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Introduction
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Tracheal tumors are rare, with vascular tumors accounting for less than 10% of these lesions [1]. Tracheal hemangioma is a benign vascular lesionprincipally characterized by being an infant pathology that regresses steadily; in adults tracheal hemangioma is exceptional [2–3]. Hemoptysis is one of the most alarming manifestations of tracheobronchial tree diseases and, although most are self-limited, they can occasionally jeopardize the patient’s life [4]. In such emergency situations the classic treatment of choice is an operation, although it does involve a high rate of morbidity and mortality.
Remy and colleagues [5] presented the first publication on bronchial embolization in the treatment of hemoptysis in 1974, since which time it has been used on occasions, especially in patients with a high surgical risk in whom conservative measures have failed [4, 6]. However, there is no experience when the origin is tracheal, caused partly because of the infrequency of this pathology [7], although embolization for bronchopulmonary pathology has laid the foundations for it to be applied to this pathology.
We report a tracheal capillary hemangioma in an adult, which began as a massive hemoptysis and was treated successfully with embolization by interventional radiology.
A 66-year-old woman was admitted with a diagnosis of idiopathic thrombopenic purpura, which responded poorly to medical treatment with corticoids and immunoglobulins, for which reason a splenectomy was indicated. On the second postoperative day she presented with bleeding limited by the nasogastric tube, for which a gastroscopy was done with normal results, and a blood test was done that revealed some 100,000 platelets/µL. Twenty-four hours later she presented with a new episode of bleeding, this time suggesting hemoptysis; at that time a bronchoscopy was performed, which showed an irregular excrescent mass with an infiltrating appearance stenosing 30% to 40% of the lumen between the first and third tracheal rings from which biopsies were taken. Twenty-four hours later she presented with a massive hemoptysis that required transfusion of blood concentrate, platelets, and plasma; the bleeding area was tamponed by balloon of endotracheal tube of the mechanical ventilation means of a bronchoscopy, leaving the patient stable but requiring multiple transfusions of platelets caused by a severe thrombopenia. A computed tomographic scan was performed that confirmed the tracheal tumor, but did not reveal infiltration or spread. Histology of the biopsy taken by bronchoscopy informed of capillary hemangioma.
The tamponade by endotracheal tube was removed at 48 hours; active bleeding was observed through the hemangioma. An arteriography was indicated and revealed a left bronchial artery forming a common trunkwith a right supreme intercostal artery from which a pathologic artery branched out toward the tracheal tumor area (Fig 1).
This branch was catheterized selectively and embolized with 300 µ to 500 µ spherical microparticles (Contour Emboli, San Francisco, CA; Boston Scientific, Natick, MA; Target Therapeutics, Fremont, CA) until arterial flow was arrested (Fig 2).
The patient was kept sedated for 24 hours with orotracheal intubation and insufflation of the balloon of the orotracheal tube at the hemangioma; the patient was later extubated without signs of bleeding. During her hospital admission she persisted with the thrombopenia that required several transfusions of platelets, resulting in a figure of 45,000/µL on discharge.
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Fig 1. Pre-embolization arteriography. Normal left bronchial artery forming a common trunk with a right supreme intercostal artery, from the apex of which a pathologic artery branches out toward the tracheal tumor area (the arrowheads mark the path of this pathologic artery).
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Fig 2. Postembolization arteriography. After embolization of the pathologic right intercostal bronchial trunk, the arterial flow along this branch is seen to be arrested (arrowhead).
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Control bronchoscopy was delayed for 5 months because of a severe thrombocytopenia requiring high doses of corticoid therapy to bring it under control; the bronchoscopy was normal. One year after the operation the patient is asymptomatic, and the control fibrobronchoscopy was found to be normal.
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Comment
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Capillary hemangiomas of the tracheobronchial tree are extremely rare in adults [2–3], and unlike cases in children, which are characterized by features of obstructive respiratory insufficiency, the most common symptoms are coughing and hemoptysis, as shown by Strausz and Soltesz [2] and Brown and Rebeiz [3]. Chest radiography is usually normal, and bronchoscopy is fundamental for diagnosis and for taking specimens for histologic confirmation, as in our case. Surgical excision is the curative treatment of choice, although there are several therapeutic alternatives such as systemic steroids, local injection of sclerosing substances, cryotherapy, laser, and radiotherapy [2–4, 7].
A therapeutic option not used up until now is embolization by means of interventional radiology of the tumor vascularization. However, recent advances in interventional radiology techniques, together with the development of emboligenic agents, have made endovascular embolization an important adjuvant or definitive treatment for multiple vascular lesions of the head, neck, and chest [4, 8]. In recent years, bronchial embolization has proved to be efficient in cases of menacing hemoptysis with contraindication for operation, or before an operation to control the hemorrhage so that the patient is in a better condition to be operated on [9]. In Spain there is currently a therapeutic guideline for the management of menacing hemoptysis [4] that describes the indications for performing a bronchial arteriography and possible embolization. These are the guidelines that enabled us to indicate embolization in our case despite it being tracheal.
The results reported for embolization in nonneoplastic vascular lesions are excellent, as in our case, with improvement of symptoms and return of physiologic parameters to normal [10]. However, it does have its risks, and one must be very careful when performing it so as not to release emboli that may affect nearby vascular structures (carotid, ophthalmic, and so forth) [10]. Therefore it is fundamental to choose a good material for embolization. One must avoid liquid solutions and particles less than 250 microns in size caused by the major risk of spread through neighboring vascular structures and causing medullary lesion or tissular necrosis [4], or both. The use of coils or occlusive balloons is not recommended either, as this only causes proximal occlusion of the vessel with the possible formation of collaterals that re-channel the distal bed [4].
In the cases described of embolizations for hemoptysis, it is brought under control in more than 90% of patients [7–8], with its major limitation being the high rate of early recurrence (14% to 20%) [6, 9, 11]. This is attributed to an incomplete embolization or re-channelling of the treated vessels, which means an exhaustive search must be made for all vessels potentially responsible for the bleeding, as in our case [11]. The late recurrence rate is also high (18% to 28%) [9, 11] and generally represents progression of the underlying pulmonary disease. The embolization was final in our case, because all vascularization of the hemangioma was embolized, thus achieving total remission of the same. If there is no base disease other than the hemangioma (eg, tuberculosis), it can be considered cured.
In conclusion, tracheal capillary hemangioma may produce a massive hemoptysis that can be controlled efficiently and definitively with embolization by interventional radiology.
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References
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- Sie K.C., Tampakopoulou D.A. Hemangiomas and vascular malformations of the airway. Otolaryngol Clin North Am 2000;33:209-220.[Medline]
- Strausz J., Soltesz I. Bronchial capillary hemangioma in adults. Pathol Oncol Res 1999;5:233-234.[Medline]
- Brown J.S., Rebeiz E.E. Pathologic quiz case 1: capillary hemangioma. Arch Otolaryngol Head Neck Surg 1993;119:700-702.[Abstract/Free Full Text]
- Ruiz J., Llorente J.L., Orega F.J., et al. Normativa sobre el manejo de la hemoptisis amenazante. Arch Bronconeumol 1997;33:31-40.[Medline]
- Remy J., Voisin C., Dupuis C., et al. Traitement des hemoptysis par embolization de la circulation systemique. Ann Radiol (Paris) 1974;17:5-16.
- Zhang J.S., Cui Z.P., Wang M.Q., Yang L. Bronchial arteriography and transcatheter embolization in the management of hemoptysis. Cardiovasc Intervent Radiol 1994;17:276-279.[Medline]
- Bailey C.M., Froehlich P., Hoeve H.L. Management of subglottic haemangioma. J Laryngol Otol 1998;112:765-768.[Medline]
- Remonda L., Schroth G., Caversaccio M., et al. Endovascular treatment of acute and subacute hemorrhage in the head and neck. Arch Otolaryngol Head Neck Surg 2000;126:1255-1262.[Abstract/Free Full Text]
- Bustamante M., García Valtuille R., Agüero R., et al. Embolización bronquial en el tratamiento de la hemoptisis. Arch Bronconeumol 1998;34:479-483.[Medline]
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- Hayakawa K., Tanaka F., Torizuka T., et al. Bronquial artery embolization for hemoptysis: immediate and long-term results. Cardiovasc Intervent Radiol 1992;15:154-159.[Medline]
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Abstract
Introduction
