INTRODUCTION: Lobular Capillary Hemangioma (LCH), formally known as pyogenic granuloma, usually present as a painless, bleeding mass adherent to the mucosal or cutaneous surfaces of the upper airways.
CASE PRESENTATION: A 22 year old male presented with recurrent episodes of hemoptysis over a 4 week period with 3 distinct events: 2-4 tablespoons of blood. Initially thought related to epistaxis, he was sent home with treatment for allergic rhinitis. He had a past medical history of eosinophillic colitis where he was on and off high-dose Prednisone, an aneurismal bone cyst status post removal, and recently diagnosed hypogonadism on injectable supplementation. He had no inhalant exposure or trauma and a family history of a mother who had vascular ectasias in her spinal cord. He did note that these episodes seemed to occur 5 days after his dose of intramuscular testosterone. Physical exam, labs, and CT were unremarkable. Upon flexible bronchoscopic evaluation, a lesion was seen in the distal trachea. The purple, vascular lesion was approximately 1-1.5 cm in size, located 3 cm from the carina at the 5 o’clock position and along the posterior wall attached by a short pedicle. A biopsy was taken at that time which only showed fibrin products and inflammatory cells. Hemostasis was obtained with use of cold saline, several injections of topical epinephrine, and Argon Plasma Coagulation. Patient was then taken to the operating suite and a therapeutic flexible bronchoscope was used with electrocautery loop snare. The lesion was obtained by endoscopic basket and sent for pathology where the diagnosis of LCH was made.
DISCUSSION: LCH is a well known entity that commonly involves skin and oral/nasal mucosa, developing rapidly over a number of days to weeks. Its cause is not well defined but certain postulates correlate to previous trauma, hormonal shifts, viral oncogenes, Bartonella infection, production of angiogenic factors, and cytogenetic clonal deletion abnormalities. Other causal relationships suggest certain drugs can also cause these lesions, such as retinoid therapy and others. Our patient had recently been started on testosterone therapy which could be coincidental or have influenced the necessary hormonal shifts to promote such a vascular lesion in a susceptible host. These lesions have commonly and extensively been studied in late pregnancy, arising mostly in the airway mucosa from the influence of female hormones on vascular development. Though the relationship between testosterone and angiogenesis has been correlated in bench studies, there has yet to be specific research regarding its role in this particular type of tumor in this instance. Also of note is that our patient had a previous history of a vascular lesion in his tibia that had been removed at the age of 16, requiring surgery and an oral endotracheal intubation suggesting a instance of remote trauma. The position of the lesion made removal technically difficult due to the posterior wall location and the vascular structure of the tumor itself. The treatment in general for these lesions depends on the extent, age, comorbidities, and other factors contributing to the overall clinical scenario. Because of inherent thinness of the posterior tracheal wall, there is a risk of iatrogenic tears and perforation. These defects are treated with various approaches; surgical intervention is most times required. LCH, depending on site and situation, have been successfully treated with various methods involving cryotherapy, YAG laser therapy, topical or intralesional steroids or neoplastic agents, propranolol in children, and surgical excision. If bleeding is a potential risk, such as it is with vascular tumors, having interventional bronchoscopy, perhaps rigid bronchoscopy, available during the removal would be highly suggested. The lesion in this case was easily removed without rigid bronchoscopy and hemostasis was controlled due to the small pedicle size.
CONCLUSIONS: LCH found in the trachea continues to be a rare entity and has yet to be described abutting the posterior wall. Posterior laryngeal wall lesions can be challenging due to risk of laceration and perforation and the appropriate equipment needs to be readily available in case of laceration. Though no direct causal link could be found in this patient, we believe that hormonal supplementation may have played a role in its development.
Reference #1 Mills SE, Cooper PH, Fechner RE. Lobular capillary hemangioma: the underlying lesion of pyogenic granuloma. A study of 73 cases from the oral and nasal mucous membranes. Am J Surg Pathol. Oct 1980;4(5):470-9.
Reference #2 Irani S, Brack T, Pfaltz M, Russi EW. Tracheal lobular capillary hemangioma: a rare cause of recurrent hemoptysis. Chest. Jun 2003;123(6):2148-9.
Reference #3 Chawla M, Stone C, Simoff M. Lobular capillary hemangioma of the trachea: the second case. J of Bronchology and Interventional Pulmonology. Jul 2010; 17(3):238-240.
DISCLOSURE: The following authors have nothing to disclose: Amy Ford-Turner, Praveen Chenna, Enrique Diaz-Guzman
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