Volume 2, Issue 4 , Pages 231-234, December 2007
Pediatric lobular capillary hemangioma accompanied with a foreign body in the nasal cavity
Article Outline
Summary
Lobular capillary hemangioma (LCH) is a hyperplastic lesion that frequently occurs due to trauma or hormonal factors either on skin or mucosal membranes. Mucosal lesions occur frequently in the oral cavity in the head and neck. Although LCH can occur in all ages, very few are reported in infants and children. Intranasal localization is very rare in this age group. We present a 12-year-old child with an intranasal mucosal LCH accompanied with a nasal foreign body. To our knowledge, our case is the first case that an intranasal LCH accompanied a foreign body in the nasal cavity.
Keywords: Lobular capillary hemangioma, Pyogenic granuloma, Nasal foreign bodies, Nasal cavity, Endoscopic surgical procedures
1. Introduction
Lobular capillary hemangioma (LCH) was first defined as “human botryomycosis” and later called as pyogenic granuloma, telangiectatic granuloma, granuloma pedunculatum or infected granuloma [1]. They are pedunculated, benign and very fast growing vascular lesions that usually involve the skin and oral mucosa, and are well known to dermatologists and oral surgeons [2]. Their etiology is not thoroughly known. It is reported that microtrauma and hormonal factors may play a role in development of those lesions. LCH's that appear during pregnancy are called “pyogenic granuloma gravidarum” and are related with hormonal changes.
Although may occur in all age groups, LCH is more frequent in women in the third and fourth decades of life. It occurs more in males than in females in infants and children [2], [3]. Very few intranasal LCH were reported in children. Katori and Tsukuda reported that intranasal LCH's were reported only in six children in English literature between 1985 and 2005 [2]. To our knowledge, an intranasal LCH and an accompanying nasal foreign body was not reported previously in English literature. In this article, we present a child with LCH and a foreign body in the nasal cavity.
2. Case report
A 12-year-old girl came to our clinic with complaints of right-sided nasal blockage and recurrent epistaxis. She reported that the nasal blockage existed for 3 months, it was progressive and she had epistaxis in the last 2 months. She had a nasal trauma 3 months ago, however she did not have any medical or surgical treatment. Her epistaxis was stopped by the patient herself or by her parents with the aid of paper handkerchiefs and external nasal compression.
Anterior rinoscopy revealed a pink-purple coloured fragile mass that completely filled the right nasal cavity and extended out of the nasal vestibule. Inferior and middle turbinates could not be seen. The right tympanic membrane was centrally 3
mm×3mm perforated. Oral cavity was normal. There was no palpable mass or lymphadenopathy in the neck.
Nasal endoscopy revealed that the mass was quite fragile, and completely filled the right nasal cavity. Inspection of the lateral nasal wall was not possible. The left nasal cavity, left lateral nasal wall, and left side of the nasal septum were normal. A lymphoid tissue (adenoid vegetation) which caused partial obstruction of the choana was present in the nasopharynx.
Axial and coronal sections of computerized tomography showed a mass of 20mm×11mm that caused thickening of the septal mucosa anteriorly. The mass obliterated the right nasal cavity and a mucosal thickening in the right maxillary sinus floor was observed (Fig. 1).
Fig. 1.
The mass in the right nasal cavity on computed tomography.
The patient was operated under general anaesthesia. By a 0 degree Hopkins telescope it was seen that the mass had a thin peduncle that originated from the anteroinferior nasal septum (Video 1). The 20mm×10mm×8mm sized mass was totally excised subperichondrially with normal mucosal margins under endoscopic view. There was no destruction or erosion in the septal cartilage. Following the resection of the mass, a greyish-white, soft, 18mm×15mm sized foreign body resembling a paper handkerchief was seen filling the space between the middle and the inferior turbinates (Fig. 2). It eroded the neighbouring septal mucosa. Foreign body was removed and sinus pack tampons with antibiotic ointment were placed in the nasal cavity. The patient did not have any postoperative complications, and she was discharged on the second postoperative day after removal of the nasal packings.
Fig. 2.
Endoscopic view of the foreign body.
The histopathological examination showed that the mass was ulcerated and was covered widely by fibrin and necrotic exuda. Coils of capillaries that were lined with young endothelial cells rested in a loose edematous stroma with infiltration of the inflammatory cells (Fig. 3). The diagnosis was “mucosal LCH”.
Fig. 3.
Coils of capillaries that were lined with young endothelial cells are visible (H&E, X 250).
Neither recurrence nor any residual disease was seen in endoscopic examination in the postoperative eighth month.
3. Discussion
LCH is a benign, fast growing vascular tumour that usually develops on skin and mucosal membranes. Previously it was named as pyogenic granuloma, however, later it was realized that this lesion was in fact neither a granulomatous reaction nor a bacterial infection. Due to histopathological findings, these lesions are defined by Mills as “lobular capillary hemangiomas” [3], [4], [5]. LCH develops fast and frequently becomes ulcerative in early phases. Mucosal LCH is mostly seen in the gingiva, lip, tongue, and buccal mucosa. Intranasal localization is very rare [3], [5].
Although LCH can occur in all ages, very few are reported in infants and children. Intranasal LCH is even rarer in this age group. El-Sayed and Al-Serhani reported that none of the 12 LCH patients were children, and only two of them were young adults [6]. In a clinicopathological research performed on 178 children with LCH, it was reported that 62% of the lesions were located in the head-neck area, 21,8% were present in mucosal membranes such as the oral cavity and conjunctiva, and only one case had LCH in the nasal cavity mucosa [7].
The mechanism of the development of these vascular tumours is still not known. It is suggested that trauma, hormonal changes, viral oncogenes, microscopic arteriovenous malformations, and angiogenic growth factors may have a role in pathogenesis [1]. Nasal packings and digital trauma must be noted among traumatic factors. Frequent localization of the LCH in Little area, in anterior part of the nasal cavity and on anterior tips of the inferior turbinates supports the role of the trauma in the pathogenesis [1].
The most frequent symptom is unilateral nasal bleeding. Nasal blockage and smell disorders may occur. Pain is not a frequent. Usually a red-pink coloured, hypervascularized, fragile, irregular, and pedunculated mass is seen in the nasal examination [1], [5]. Our case had complaints of nasal blockage and recurrent epistaxis in the right side of the nose. It was interesting to see that the complaints started following trauma, and that after a short time the mass enlarged so much that it extended out of the nasal vestibule. In the endoscopic examination it was observed that the mass was hypervascular, pedunculated and originated from the Little area on the right side of the nasal septum. All of those findings suggested a traumatic etiopathogenesis in our case and this finding is in agreement with the literature.
Nasal tampons are frequently used for epistaxis and for postoperative control of the nasal bleeding. Vaseline gauze, inflatable balloons, and merocel are widely used. Vaseline gauze tampons are very rough, traumatic and cause discomfort [5]. There are four cases that developed LCH after the use of gauze tampons [5], [8], [9], [10]. Although our patient and her family expressed that the patient did not have any medical or surgical intervention after the trauma, they told that they tried to stop repeated nose bleeds with compress and tissues. The presence of a foreign body (paper handkerchief) behind the mass suggests that long standing irritation of the nasal mucosa may result in the formation of the LCH.
Histological findings are characterized by the polypoid, circular, exophytic, and lobular proliferation of capillaries in a fibromyxoid stroma [1]. Toida et al. reported that discrimination of lobular and superficial ulcerative areas was possible in most of the cases [11]. Lobular area shows characteristic lobular proliferation of microvascular elements and the ulcerative area shows superficial neutrophilic infiltration and irregular dilatation of blood vessels [1], [3]. The mass of our case was ulcerated and was covered widely by fibrin and necrotic exuda. There were coils of capillaries that were lined with young endothelial cells in a loose edematous stroma with infiltration of the inflammatory cells.
Congenital, inflammatory, and neoplastic nasal masses must be considered in differential diagnosis: polyps, sarcoidosis, Wegener granulomatosis, meningoencephalocele, glioma, fibroma, nasopharyngeal cyst, lipoma, hemangioperistoma, histiocytoma, leiomyoma, osteoma, squamous cell carcinoma, adenocarcinoma, esthesioneuroblastoma, and angiosarcoma [12]. It must be kept in mind that the foreign body can mimic an intranasal mass in children.
The treatment of LCH is surgical excision. Endoscopic surgery is the preferred approach for big lesions that fill the nasal cavity completely and extend into the nasopharynx as well as for small lesions [1]. Endoscopic surgery enables perfect examination of the lesion and adjacent tissues. Optimal bleeding control can be performed because of good visualization of the surgical area. The main factor of obtaining successful results is the resection of the mass subperichondrially or subperiostially with normal mucosal margins [1]. In our case, the mass was excised subperichondrially with normal mucosal margins. Surprisingly, a foreign body resembling a paper handkerchief was seen behind the mass after its removal. The foreign body seemed infected and it caused maceration of the septal mucosa. We did not encounter any recurrence or residual mass in the postoperative period.
To our knowledge, there are no reports in the literature concerning simultaneous intranasal LCH and an intranasal foreign body either in adults or in children. In patients of the pediatric age group who suffer from unilateral nasal blockage and epistaxis, it must be kept in mind that a foreign body may accompany LCH. Therefore, following resection of the mass, the nose must be examined for the presence of any foreign bodies with the aid of nasal endoscopes and those must be removed to prevent any recurrences.
Appendix A. Supplementary data
Video 1. Endoscopic resection of the mass and the foreign body.
References
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PII: S1871-4048(07)00057-3
doi:10.1016/j.pedex.2007.06.009
© 2007 Elsevier Ireland Ltd. All rights reserved.
Volume 2, Issue 4 , Pages 231-234, December 2007
