Lupine Publishers | Lobular Capillary Hemangioma of Head and Neck Region: A Retrospective Study in A Tertiary Centre

 Lupine Publishers | Journal of Otolaryngology

 

Abstract

Background: Lobular capillary hemangiomas (LCH) are uncommon benign vascular entity of unknown etiology. Their prime locations are skin and mucosa of oral cavity, however, the nasal cavity involvement is very rare pertaining to head and neck vascular lesions. Here, we have retrospectively analyzed the confirmed LCH cases including sites, clinical features, radiological findings, treatments, and histological results.

Materials and Methods: A retrospective study carried out in Department of Ear, Nose and Throat and Head and Neck Surgeries (ENT-HNS) of Universal College of Medical Sciences, Tribhuvan University Teaching Hospital (UCMS, TUTH), Bhairahawa, Nepal. All the data of 10 cases (from June 2014 to February 2020) were collected from the OT (operation theatre) register and hospital records section. Data were analyzed for symptoms, possible etiologic factors, demographic profile, CT findings, histopathological features, and treatment modalities.

Results: Unilateral nasal obstruction with nasal mass and intermittent epistaxis (90%) were the chief complaints. Female predominance was favored. In 80% of the patients, the mass was arising from the anterior nasal septum, in 10% from the anterior end of the inferior turbinate and in 10% from the upper lip. GA (general anesthesia) was used in 20% of cases whereas LA (local anesthesia) was used in 80% of the cases. All patients were managed successfully with no recurrence at follow up of 6 months. Conclusion: It is a rare benign vascular entity with slight female predominance. It bears a less morbidity and recurrences on surgical excision but should be differentially diagnosed rightly so as to minimize aggressive management.

Keywords: Capillary hemangioma; nasal cavity; vascular lesion

Abbreviations: LCH: Lobular Capillary Hemangiomas; LA: Local Anesthesia; GA: General Anesthesia; ENT-HNS: Ear, Nose and Throat and Head and Neck Surgeries; OT: Operation Theatre; HPE: Histopathology Evaluation

Introduction

Lobular capillary hemangioma (LCH) is synonymously known as pyogenic granuloma, epulis gravidarum, eruptive hemangioma, granulation tissue-type hemangioma, granuloma gravidarum, pregnancy tumor and botryomycome [1-3]. It is a benign, fast growing, capillary proliferation with a microscopically distinct lobular architecture that affects the skin and mucous membranes of the oral and, rarely, of the nasal cavities and internal organs such as brain and liver [4-9]. Poncet and Dor were the first ones to describe it in 1897 as human botryomycosis and referred to these tumors as small vascular tumors in the fingers of four patients [1,7,10,11]. Oral cavity mucosa is the predominant site for LCH occurrence, but nasal cavity involvement is rare. Anterior septum (Little’s area) is the most frequently affected site followed by turbinate, but lesions have also been described arising from the maxillary sinus, roof of nasal cavity and floor of nasal vestibule [3,7,8]. The exact pathogenesis seems to be debatable but it’s linked with microtrauma (nose picking or nasal packing resulting in the overgrowth of granulation tissue) and hormonal factors such as pregnancy and oral contraceptive use as possible etiologies and other plausible being viral oncogenes, microscopic arterial venous malformations and over production of angiogenic growth factors [3-7]. Patients usually present with painless intermittent epistaxis, progressive unilateral nasal obstruction, and nasal mass of a short period. There’s equal distribution between men and women in all ages however some studies show female predilection in the third to fifth decades [7,8,12-14]. The aim of this retrospective study is to analyze the clinical presentation, histological and radiological findings, and the treatment strategy.

Materials and Methods

This is a retrospective study carried out in Department of Ear, Nose and Throat and Head and Neck Surgeries (ENT-HNS) of Universal College of Medical Sciences, Tribhuvan University Teaching Hospital (UCMS, TUTH), Bhairahawa, Nepal. All the data (from June 2014 to February 2020) were collected from the OT (operation theatre) register and hospital records section. Institutional ethical clearance has been obtained. Meantime patients were informed about the future possibility of paper publication and the use of their hospital records use and the pictures too.

Inclusion Criteria

All patients with confirmed HPE (histopathology evaluation) of LCH of the nose.

Exclusion Criteria

Patients with bleeding diathesis, and other comorbidities. Information regarding symptoms, possible etiologic factors, demographic profile, CT findings, histopathological features, and treatment modalities were reviewed. All the cases were done in the OT using either general or local anesthesia. All cases underwent surgical excision and electrocauterization of the base and sent for histopathological evaluation. Zero-degree rigid endoscopy was used in 3 cases and headlight was used in 7 cases. After the anesthetization of the case, 5 ml of 2% Xylocaine with 1:200000 Adrenaline was locally infiltrated. Ribbon gauze packs impregnated with Oxymetazoline nasal drops and 5ml of 4% Xylocaine were kept for 10 minutes then the procedure was performed. Soframycin ointment impregnated ribbon gauze pack were kept for 1 day unilaterally. Patients were discharged on the same day on oral Ciprofloxacin and analgesics. On next day, the nasal pack was removed in the OPD and topical Mupirocin ointment was prescribed for 2 weeks.

Results

There were total of 10 cases. 2 (20%) were male and 8 (80%) female patients (table 1) with male to female ratio of 1:4. The mean age was 30.6 years, ranging from 18 to 57 (Table 1) 1 male and 3 females were in the 5th decade. 2 female patients had a history of oral contraceptives intake. 1 male and 4 females had history of frequent nose picking habit. In 9 patients, the chief complaints were unilateral rapidly growing nasal mass, intermittent epistaxis and nasal obstruction and rhinorrhea in 1 patient. In 1 male patient (18 years old) the complaint was rapidly growing painless growth in the upper lip. In 1 female patient in the 5the decade the mass was arising from the inferior turbinate (left side) (Figures 1-6) and the rest being from the anterior end of nasal septum (6 on the right side and 2 on the left). The mean duration of symptoms was 2-3 months in the majority however 1 female patient in the 5th decade was having for 6-8th months and 1 male patient had it since childhood (Table 1). Endoscopic removal was used in 3 cases and 7 were dealt with headlight. On examination, the mass ranges from 1.5 – 2.5 cm in size and was single dark red to polypoidal which bleed easily on brisk touch by the instrument. CT was suggestive of well-defined, enhancing soft tissue density lesions without bony erosions. Maxillary mucosal thickening (ipsilateral) was found in 1 patient (13.3%). There were no postoperative complications with 1 recurrence case who lost to follow up till date (Table 1). Histopathology showed section of ulceration and granulation with underlying stroma of increased proliferation of small calibered blood vessels lined by plump of flattened endothelial cells; few in staghorn pattern and vaguely arranged in lobules. Intervening stroma is fibro collagenous and also shows loose, edematous areas along with mixed inflammatory cells comprised of lymphocytes, plasma cells, and neutrophils. Areas of necrosis is also evident. No atypia.

Figure 1: A red colored polypoidal mass in left nasal cavity.

Figure 2: Coronal CT showing heterodense soft tissue density in left inferior turbinate.

Figure 3: Axial CT showing soft tissue density in left nasal cavity.

Figure 4: Endoscopic view of left nasal mass attached to left inferior turbinate.

Figure 5: In toto excised mass 3x2.5x2.5 cm.

Figure 6: Hematoxylin & eosin stained picture of capillary lobular hemangioma.

Table 1: Demographic characteristics of patients with lobular capillary hemangiomas of the head and neck region.

Discussion

Frank and Blahd M first described LCH as pyogenic granuloma or bleeding polypus in 1940. Again, due to its characteristic microscopic features Mills et al termed pyogenic granuloma as LCH [15]. No malignant transformation has been reported till date in these kinds of benign tumors [7]. As per the literature, LCH is predominant in females and most commonly occur in the third decade of life [5]. In our series, although two-thirds of the patients were female, supports the idea of higher incidence in the third decade (60%). The mean age was 30.6 years, ranging from 18 to 57. 1 male and 3 females were in the 5th decade. 2 female patients had a history of oral contraceptives intake. 1 male and 4 females had history of frequent nose picking habit. In 9 patients, the chief complaints were unilateral rapidly growing nasal mass, intermittent epistaxis and nasal obstruction and rhinorrhea in 1 patient. In 1 male patient (18 years old) the complaint was rapidly growing painless growth in the upper lip. In 1 female patient in the 5the decade the mass was arising from the inferior turbinate (left side) and the rest being from the anterior end of nasal septum (6 on the right side and 2 on the left). The mean duration of symptoms was 2-3 months in the majority however 1 female patient in the 5th decade was having for 6-8th months and 1 male patient had it since childhood.

Narayanaswamy et al. (2015) study also showed 80% (16/20) of females and 80% of the study population were in the third decade [7]. 10 had a history of oral contraceptive pills intake. Of the 4/20 male patients, 2 were in the first decade, and 2 in the third decade. Main symptoms were a unilateral nasal obstruction (80%) and epistaxis (60%) which was unprovoked or with trivial trauma. In 14/20 (70%) patients the mass was seen arising from the caudal end of the septum, in 4 patients (20%) from the anterior end of the middle turbinate and in 2 patients (10%) from the anterior end of inferior turbinate. Chi et al. (2014) study also showed female predominance 10/15 but no peak incidence was found in the third decade [8]. 3/15 had diabetes mellitus, hypertension, and rheumatoid arthritis. All patients presented with unilateral nasal complaint with 60.0% (9/15) of the lesions on the left side and 40.0% (6/15) on the right. Main symptoms were epistaxis (14/15, 93.3%), followed by nasal obstruction (12/15, 80.0%), rhinorrhea (8/15, 53.3%), protruding tumor (3/15, 20.0%), facial pain (2/15; 13.3%), and headache (1/15; 6.7%). The mean duration of symptoms prior to admission was 3.4±3.9 months (ranging from 2 weeks to 12 months). The most common site of the lesion was the anterior nasal septum (6/15, 40.0%), followed by the inferior turbinate (4/15, 26.6%), nasal vestibule (3/15, 20.0%), posterior nasal septum (1/15, 6.7%) (Figure 1), and middle turbinate (1/15, 6.7%). However, Puxeddu et al. study (2006) did not support the idea of female predominance and also the peak incidence being in the fifth decade of life. Predisposing factors such as nasal trauma and pregnancy were identified in 6 (15%) and 2 (5%) patients, respectively. Presenting symptoms were unilateral epistaxis (95%), nasal obstruction (35%), rhinorrhea (10%), facial pain (7.5%), headache (4%), and hyposmia (4%) alone or in different associations and the duration being 1 week to 5 years [9].

In all the study, CECT (contrast enhanced computer tomogram) showed well enhanced soft tissue without bony erosions. The mass was excised via surgical excision and electrocautery either classically with the head light or endoscopically in all the study. Surgical excision is the preferred treatment modality for lobular capillary hemangioma of the nasal cavity. Various surgical treatment modalities include electrocoagulation, cryotherapy, LASER, excisional surgery, and excisional surgery following angiography with embolization (8). In our study, the mass was excised via surgical excision and electrocautery either classically with the head light or endoscopically without preoperative embolization. The recurrence rate ranges from 0% to 42.0%, depending on the case series and the duration of follow-up [8]. Recurrence was not seen in none of the series. The differential diagnoses of such mass includes nasal polyp, antrochoanal polyp, meningocele, meningoencephalocele, papilloma, wegner’s granulomatosis, sarcoidosis, Kaposi sarcoma, hemangiosarcoma, squamous cell carcinoma and mucosal malignant melanoma and in children congenital malformations, dermoid cyst, angiomatous polyp, meningocele, angiofibroma, glioma and schwannoma should be kept in mind. Biopsy of the mass is not recommended by any study due to its vascular origin and high risk of nosebleed.

Conclusion

LCH is quite a rare benign entity in the nasal cavity region. There is a female predominance with debatable etiology and usually the presenting symptoms are unilateral progressive nasal obstruction and epistaxis of shorter duration. Keeping the differential diagnoses in mind, surgical excision is the definitive treatment modality with less recurrences and minimal morbidity. Awareness of such lesions may help in misdiagnoses of more aggressive entities.

Conflicts of Interest

The authors declare they have no potential conflicts of interest.

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Lupine Publishers | Detection of Supernumerary Tooth in the Nasal Cavity

 Lupine Publishers | Journal of Otolaryngology

Abstract

Aims and objectives: To detect super numeracy tooth in nasal cavity.

Introduction: The ectopic dentition present in intranasal area is very rare finding clinically. It is very difficult to make proper diagnosis of such condition. Teeth is present in ovaries, testes and pre-sacral regions. The presence of teeth in maxillary sinus, mandibular condyle, chin, nose and orbit in maxillofacial region is also observed.

Case Report: Prospective cross-sectional study was conducted. Anterior microscopy was done; proper detection of supernumerary tooth was carried out.

Result: A 21-year-old female patient presented with complaints of intermittent right sided epistaxis. She had history of occasional headache for 6 months. An anterior rhinoscopic examination revealed a hard white-mass which is peripherally surrounded by granulation tissue that lies in floor of the right-nasal cavity. It’s appearance clinically was of a rhinolith.

Conclusion: Super-numeracy teeth are observed extra to normal complement indentation. They may occur unilaterally or bilaterally, single or multiple. A variety of complications can occur that may be crowding or cyst formation. There by, identifying early and also treatment are useful for management.

Keywords: Supernumerary; ectopic; dentition; Epistaxis; Rhinoscopic examination

Introduction

Ectopic dentition situated in intranasal region is very rare thing which is clinically observed. The proper measures should be adapted to detect supernumerary tooth. Teeth are present in ovaries, anterior mediastinumandpresacral regions, testes [1,2], also found in maxillary- sinus, mandibular-condyle, coronoid-process, chin, nose and orbit in the maxillofacial region, while rarely seen in intranasal region. Most common conditions which are associated with a raised prevalence of ectopic teeth are cleft lip and palate, cleido-cranial dysplasia and Gardner syndrome. Intra-nasal teeth can lead to problems e.g. Nasal-obstruction, chronic-rhinorrhoea and speech-defects. Most common ectopic-tooth which arises in the maxillary midline is known as a mesiodens. This unusual condition should be supposed to be present in patients with nasal blockage/ obstruction and unilateral purulent rhinorrhoea [3-7]. So, this study was conducted to detect the supernumerary tooth in nasal cavity.

Case Report

A twenty-one-year-old female patient presented with complaints of intermittent right sided epistaxis and history of occasional headache for 6 months. An anterior rhinoscopic examination revealed a hard- white mass surrounded by granulation tissue lying in the floor of the right nasal cavity. The clinical appearance was that of a rhinolith. An orthopantomography revealed the presence of a radio-opaque tooth shape mass, suggestive of a supernumerary tooth since the intraoral dental occlusion was complete. Computed Tomography (CT) scan was required to assess the exact position of the presumed-supernumerary tooth and its relationship with the surrounding structures. The CT examination showed that the mass lesion was like a conical shaped structure tapering to a point suggestive of a tooth lying in the anteroposterior direction (Figure 1). The patient had gone an endoscopic removal of the supernumerary tooth under general anesthesia. Once tooth was localised, the covering periosteum was removed, location of tooth was deviated from its area of impaction and it was removed with forceps from the right nostril (Figures 2 & 3). Uneventful postoperative course was observed.

Figure 1: Clinical appearance on anterior rhinoscopy

Figure 2: CT scan showing relation if tooth with surrounding structures

Figure 3: Supernumerary tooth after removal

Discussion

It is observed that 0.1 to 1 percent of population is influenced by and having supernumerary tooth, with most common situation as upper incisor area called as mesiodens. The causes of supernumerary teeth are not totally understood. According to one theory, supernumerary tooth is generated either from a thin tooth bedwhicharisesfrom the dental lamina close to permanent tooth bud /or from division of the permanent bud itself [1,2]. While another theory states that their development is due to reversion of dentition of primates that are extinct and had 3 pairs of incisors. There is formation of supernumeraries due to local, independent, conditioned hyperactivity of dental lamina, as suggested by hyperactivity theory. There is a role of heredity also, because supernumeraries are commonly seen in relatives of children who are affected as compared to general population. But, simple Mendelian pattern is not followed by anomaly [3-5]. The reason of this ectopic growth formation in not clearly understood, it hasbeen attributed to obstructionatthe time of tooth eruption secondary to crowded dentition, persistent deciduous teeth/or exceptionally dense bone. Some pathogenic factors responsible are genetic predisposition, developmental disturbances, rhino genic or odontogenic infection and displacement as a result of trauma/ cysts [2-4]. Various supernumerary teeth are not seen in individuals with no other associated diseases/syndromes. It is usually related with cleft lip and palate, cleidocranial dysplasia and Gardner syndrome whereas there is no significant sex distribution in primary supernumerary teeth [4-7]. The excess teeth have an atypical crown which is vertical, horizontal /inverted position. They may grow and appear on the palate as extra teeth/they may grow in the nasal cavity. The teeth may be asymptomatic or cause a variety of signs and symptoms. Complications of nasal teeth are rhinitis caseosa with perforation, aspergillosisandoronasal fistula [2,3]. The diagnosis is done on the basis of clinical and radiographic findings. Clinically, an intranasal tooth is seen as a white mass in the nasal cavity surrounded. Radiographically, it seems radiopaque lesions with the similar attenuation as that of the oral teeth. With the bone window setting, the central radiolucency, which is correlated with the pulp cavity, may have a spot or slit, depending on the orientation of the teeth. Soft tissue found in clinical and pathologic examination, surrounds the radiopaque lesion and consistent with granulation tissue [4-7]. The differential diagnosis of nasal teeth is:

a. Radiopaque foreign body,
b. Rhinolith,
c. Inflammatory lesions of syphilis, tuberculosis (or) fungal infection with calcification,
d. Benign tumours, as haemangioma, osteoma, calcified polyps, enchondroma,
e. Dermoid and malignant tumours, as chondrosarcoma and osteosarcoma.

Whereas, the Computed Tomography (CT) findings of toothequivalent attenuation and a centrally located cavity are highly discriminating features that are useful to approve the diagnosis. To remove nasal teeth, it is generally preferred to alleviate the symptoms and to avoid complications. When extra tooth is in the nasal cavity, the procedure followed is usually a minor operation. When a supernumerary tooth presents with a bony socket in the floor of the nose, its extraction may prove to be extremely challenging. CT is useful to assess the depth of site of eruption. The good time to remove the tooth is after the roots of the permanent teeth have completely formed, to prevent any injury while their development.

Conclusion

Supernumerary teeth are extra to normal complement in dentition. They may occur unilaterally or bilaterally, single or multiple. A variety of complications can occur ranging from crowding to cyst formation. Hence, early identification and treatment are essential for management.

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