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Tracheal Lobular Capillary Hemangioma*: A Rare Cause of Recurrent Hemoptysis FREE TO VIEW

Sarosh Irani; Thomas Brack; Madeleine Pfaltz; Erich W. Russi
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*From the Departments of Internal Medicine (Drs. Irani, Brack, and Russi) and Pathology (Dr. Pfaltz), Pulmonary Division, University Hospital, Zurich, Switzerland.

Correspondence to: Sarosh Irani, MD, Pulmonary Division, Department of Internal Medicine, University Hospital, CH-8091 Zurich, Switzerland; e-mail: sarosh.irani@dim.usz.ch



Chest. 2003;123(6):2148-2149. doi:10.1378/chest.123.6.2148
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Lobular capillary hemangioma (LCH) is a polypoid form of capillary hemangioma occurring on the skin and mucosal surfaces. While LCH of the oral and nasal cavity is a well-known entity, tracheal localization is extremely rare. We present the case of a 72-year-old woman with recurrent hemoptysis due to a small tumor of the proximal trachea. By endoscopic removal of the tumor by flexible bronchoscopy, the diagnosis of LCH was made, and during the following year there was no recurrent hemoptysis. To our knowledge, this is the first case of histologically proven LCH of the trachea.

Figures in this Article

Lobular capillary hemangioma (LCH) is a benign lesion that may occur either on skin or mucosal surfaces, the latter accounting for about 60% of all cases.1 LCH has a distinctive lobular arrangement of capillaries in an edematous, fibroblastic stroma. The surface is occasionally ulcerated, and the lesion may have an inflammatory cell infiltrate. The often-used term pyogenic granuloma is a misnomer, because the tumor is neither induced by bacterial infection nor is it a true granuloma.2 In a review of 639 cases of vascular lesions of the oral cavity and upper respiratory tract by Mills et al,2 73 cases with LCH were found, and none of them was localized in or below the larynx. The following case illustrates the occurrence of an LCH of the tracheal mucosa and its successful removal by flexible endoscopy.

A 72-year-old woman was in good health until she experienced an episode of cough and minor hemoptysis that lasted a few days. These symptoms recurred after an interval of 6 weeks when the patient was seen in our clinic for the first time. The patient had no history of previous intubation or other intervention in the upper airways. On physical examination, a goiter was found, but the lungs and heart were normal. The results of hematologic and chemical laboratory tests were unremarkable, and the levels of thyrotropin, free triiodothyronine, and free thyroxine were normal. A CT scan of the chest showed an asymmetric nodular enlargement of both lobes of the thyroid gland with a locally narrowed and displaced trachea. Bronchoscopy revealed a polypoid tracheal tumor, 0.2 to 0.3 cm in size, with a hyperemic overlying mucosa 3 cm below the vocal cords (Fig 1 ). The other parts of the trachea, which had a normal lumen, were unremarkable. With a flexible biopsy forceps, the tumor was completely removed, and the subsequent mild bleeding stopped a few minutes after the local instillation of adrenaline (0.1 mg/mL). Histologic examination revealed numerous capillaries arranged in a lobular pattern. The endothelial cells had a bland appearance, and occasional regular mitotic figures were present. The lobules were separated by fibrous stroma with mild accompanying inflammatory changes. The overlying epithelium was intact (Fig 2 ). There were no signs of malignancy (eg, of invading thyroid cancer), and the diagnosis of an LCH was made. During the ensuing year, the patient remained completely asymptomatic. She had no associated LCH of the skin or oral cavity.

LCH is a common polypoid form of capillary hemangioma that, aside from the skin, occurs on mucosal surfaces such as the oral and nasal cavity,2 the tongue,3 the conjunctiva,4 the duodenum,5 or the colon.6 LCH has a distinctive lobular arrangement of capillaries of various sizes in a fibromyxoid matrix. Atypical mitoses are not seen.2 The lesions often are altered by ulceration of the surface epithelium, resulting in inflammatory changes of the stroma and invasion by microorganisms. The pathogenesis of LCH is not well understood, and an infectious etiology, as is suspected by some investigators,7 has not been proven. Occasional LCHs develop following minor trauma like nasal packing8 or surgery,4 but this appears to play a role in only a minority of cases. Moreover, LCH occurs typically on the gingival surface during pregnancy (granuloma gravidarum) and may regress following delivery.

The most common presenting symptom of LCH is bleeding and/or obstructive symptoms in the case of LCH of the nasal cavity.8 Pain is not a typical feature.

Fechner and colleagues9 reviewed 62 vascular lesions from the trachea and larynx. They found no case with the typical histologic appearance of LCH. All lesions were composed of granulation tissue, and all patients had a history of trauma (ie, tracheotomies, prolonged endotracheal intubations, tracheoesophageal fistulas, biopsies, or crush injuries). Drosnes and Zwillenberg10 reported the case of a 7-year-old boy with a laryngeal tumor that was 1 cm in diameter 3 months following short-term endotracheal intubation, which initially had been thought to be an LCH. However, a review of the specimen revealed features characteristic of postintubation granuloma (also called a granulomatous polyp). Our patient had never undergone endotracheal intubation nor had she experienced another local trauma in the past. In addition, histologic appearance was typical for LCH and did not show the radial arrangement of capillaries that is seen in granulomatous polyps. To our knowledge, this is the first case of tracheal LCH reported in the literature.

For cutaneous LCH, many effective treatment modalities have been reported, including excision, curettage, electrodesiccation, chemical cauterization, and laser surgery.2 Mucosal LCH has been treated with snare cautery,6 excision biopsy,11 or plaque radiation.4 The recurrence of skin and mucosal LCH after local therapy is a well-known phenomenon; however, malignant transformation has not been reported. In our patient, the LHC was removed using biopsy forceps, and the patient remained asymptomatic subsequently. Although not conclusively proven, the lesion was most likely the source of the hemoptysis. Recurrent bleeding is a well-known characteristic of LCH, and the patient had otherwise unremarkable endoscopy and CT scan findings. Because the patient had no further symptoms, repeat bronchoscopy was not performed.

Abbreviation: LCH = lobular capillary hemangioma

Figure Jump LinkFigure 1. Endoscopic view of the proximal trachea of our patient, showing a polypoid tumor of the cartilaginous area, measuring 0.2 to 0.3 cm in size, approximately 3 cm below the vocal cords.Grahic Jump Location
Figure Jump LinkFigure 2. Pathologic specimen of our patient, showing an exophytic mass, with intact overlying epithelium and variably sized capillaries in a typically lobular arrangement, in an edematous stroma with a mild associated inflammatory infiltrate (hematoxylin-eosin, original ×80).Grahic Jump Location
Kerr, DA (1951) Granuloma pyogenicum.Oral Surg4,158-176. [PubMed] [CrossRef]
 
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 Pathol1980;4,470-479. [PubMed]
 
Sheth, SN, Gomez, C, Josephson, GD Pathological case of the month: diagnosis and discussion; pyogenic granuloma of the tongue.Arch Pediatr Adolesc Med2001;155,1065-1066. [PubMed]
 
Gunduz, K, Shields, CL, Shields, JA, et al Plaque radiation therapy for recurrent conjunctival pyogenic granuloma.Arch Ophthalmol1998;116,538-539. [PubMed]
 
Hirakawa, K, Aoyagi, K, Yao, T, et al A case of pyogenic granuloma in the duodenum: successful treatment by endoscopic snare polypectomy.Gastrointest Endosc1998;47,538-540. [PubMed]
 
Chen, TC, Lien, JM, Ng, KF, et al Multiple pyogenic granulomas in the sigmoid colon.Gastrointest Endosc1999;49,257-259. [PubMed]
 
Lee, J, Lynde, C Pyogenic granuloma: pyogenic again? Association between pyogenic granuloma and Bartonella.J Cutan Med Surg2001;5,467-470. [PubMed]
 
Lee, HM, Lee, SH, Hwang, SJ A giant pyogenic granuloma in the nasal cavity caused by nasal packing.Eur Arch Otorhinolaryngol2002;259,231-233. [PubMed]
 
Fechner, RE, Cooper, PH, Mills, SE Pyogenic granuloma of the larynx and trachea: a causal and pathologic misnomer for granulation tissueArch Otolaryngol1981;107,30-32. [PubMed]
 
Drosnes, DL, Zwillenberg, DA Laryngeal granulomatous polyp after short-term intubation of a child.Ann Otol Rhinol Laryngol1990;99,183-186. [PubMed]
 
Okumura, T, Tanoue, S, Chiba, K, et al Lobular capillary hemangioma of the esophagus: a case report and review of the literature.Acta Pathol Jpn1983;33,1303-1308. [PubMed]
 

Figures

Figure Jump LinkFigure 1. Endoscopic view of the proximal trachea of our patient, showing a polypoid tumor of the cartilaginous area, measuring 0.2 to 0.3 cm in size, approximately 3 cm below the vocal cords.Grahic Jump Location
Figure Jump LinkFigure 2. Pathologic specimen of our patient, showing an exophytic mass, with intact overlying epithelium and variably sized capillaries in a typically lobular arrangement, in an edematous stroma with a mild associated inflammatory infiltrate (hematoxylin-eosin, original ×80).Grahic Jump Location

Tables

References

Kerr, DA (1951) Granuloma pyogenicum.Oral Surg4,158-176. [PubMed] [CrossRef]
 
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 Pathol1980;4,470-479. [PubMed]
 
Sheth, SN, Gomez, C, Josephson, GD Pathological case of the month: diagnosis and discussion; pyogenic granuloma of the tongue.Arch Pediatr Adolesc Med2001;155,1065-1066. [PubMed]
 
Gunduz, K, Shields, CL, Shields, JA, et al Plaque radiation therapy for recurrent conjunctival pyogenic granuloma.Arch Ophthalmol1998;116,538-539. [PubMed]
 
Hirakawa, K, Aoyagi, K, Yao, T, et al A case of pyogenic granuloma in the duodenum: successful treatment by endoscopic snare polypectomy.Gastrointest Endosc1998;47,538-540. [PubMed]
 
Chen, TC, Lien, JM, Ng, KF, et al Multiple pyogenic granulomas in the sigmoid colon.Gastrointest Endosc1999;49,257-259. [PubMed]
 
Lee, J, Lynde, C Pyogenic granuloma: pyogenic again? Association between pyogenic granuloma and Bartonella.J Cutan Med Surg2001;5,467-470. [PubMed]
 
Lee, HM, Lee, SH, Hwang, SJ A giant pyogenic granuloma in the nasal cavity caused by nasal packing.Eur Arch Otorhinolaryngol2002;259,231-233. [PubMed]
 
Fechner, RE, Cooper, PH, Mills, SE Pyogenic granuloma of the larynx and trachea: a causal and pathologic misnomer for granulation tissueArch Otolaryngol1981;107,30-32. [PubMed]
 
Drosnes, DL, Zwillenberg, DA Laryngeal granulomatous polyp after short-term intubation of a child.Ann Otol Rhinol Laryngol1990;99,183-186. [PubMed]
 
Okumura, T, Tanoue, S, Chiba, K, et al Lobular capillary hemangioma of the esophagus: a case report and review of the literature.Acta Pathol Jpn1983;33,1303-1308. [PubMed]
 
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