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https://doi.org/10.5272/jimab.2019251.2337 Journal of IMAB Journal of IMAB - Annual Proceeding (Scientific Papers). 2019 Jan-Mar;25(1) ISSN: 1312-773X https://www.journal-imab-bg.org Case reports

PYOGENIC IN THE ORAL CAVITY: A SERIES OF CASES

Deyan Z. Neychev1, Radka B. Cholakova1, Tanya I. Sbirkova1, Stanimir N. Kisselov1, Svitlana Y. Bachurska2, Dimitar T. Atanasov1 1) Department of Oral Surgery, Faculty of Dental Medicine, Medical University of Plovdiv, Bulgaria 2) Department of General and Clinical Pathology, Medical University of Plovdiv, Bulgaria.

ABSTRACT women [1, 2]. Hullihen was the first to report such case in Introduction: Pyogenic granulomas represent tumor- 1844, and Hartzel introduced the term “pyogenic ” like lesions affecting skin and the oral cavity. This classic in 1904 [3, 4]. According to other authors, the condition is definition can be somewhat misleading because such lesion first described in 1879 by Ponect and Dor under the term is not associated with infection and lacks any clinical evi- “botryomycosis hominis” and later in 1903 Crocker pro- dence of pus or histological evidence of actual granulation posed the name “” or “Crocker and tissue. Scientific literature sources estimate its prevalence Hàrtzel disease” [2]. PG is a type of inflammatory hyperpla- to 1:25000 per capita, affecting females twice as often. Pyo- sia. This definition summarizes a number of nodular genic granuloma in the oral cavity affects the interdental overgrowths of the , histologically presented papilla in 70% of the cases. by inflammatory fibrous or granulation tissue [4]. Such Purpose: The authors present a case series of pyo- overgrowths include inflammatory fibrous genic granulomas in the oral cavity with varying localiza- (clinical , fissuratum and polyp), giant tion and therapeutic approach. cell epulis, epulis gravidarum, papillary palatal hyperpla- Materials and methods: This article presents six sia and PGhyperplasia“. clinical cases of PG. Diagnosis is challenging due to simi- Etiology of PG is yet unknown, however most asso- larities with number of tumorous and non-tumorous neo- ciated factors are trauma, , infectious agents, plasms (formations) in the oral cavity. Two histological types tartar, foreign bodies, immunosuppression, hormonal of pyogenic granuloma can be identified: lobular and non- changes, medication [2, 3, 5]. Other causes can be gingival lobular . Surgical excision is treat- inflammation due to poor and trauma due to ment method of choice, followed by deep curettage of the deciduous teeth, failure of eruption, orthodontic patho- lesion toward the underlying bone. Such precautions are logies, dental implants [2, 6]. Pyogenic granuloma is usu- necessary because 15,8% of the lesions tend to recur over ally presented as red, painless formation that is often local- time. ized in the gingiva and grows quickly, it is easily trauma- Results: Alternative therapeutic approaches for re- tized causing bleeding [3]. Other typical localizations in- moval of PG are explored, which are aimed at reducing the clude the , , and buccal mucosa [7, 8, 9]. recurrences after surgical treatment. Such opportunity is The treatment of PG is usually surgical excision, how- provided by utilization of Er:Yag laser, because its effect ever a tendency for recurrence is observed, especially in can reach the underlying bone. female patients [3]. Conclusion: Pyogenic granuloma represents a diag- nostic challenge, specifically in cases of atypical localiza- CASE PRESENTATION tion. Effective surgical approach requires complete removal Case 1: of the pathological process from the surrounding healthy A 52 years old female patient in good general health structures in order to prevent recurrences. is referred to the Department of Oral surgery of FDM – Plovdiv because of formation on the back of the tongue Keywords: Oral, pyogenic granuloma, benign vas- (Fig. 1). The patient complaints of discomfort during talk- cular tumors, Er-YAG laser, ing and eating are dated back 2-3 months. Clinical evalua- tion reveals the presence of 5-7mm large formation on the INTRODUCTION: back of the tongue that is soft and painless with palpation. Pyogenic granuloma is common, usually solitary, Periodontal status reveals tartar and plaque deposits. Treat- lobular formation of benign proliferation in the skin and ment – the formation was surgically excised and there was mucous membrane of the oral cavity. It can be pedunculated no recurrence during the 6 months follow-up period. Bi- or sessile, predominantly developing in children and young opsy showed lobular .

J of IMAB. 2019 Jan-Mar;25(1) https://www.journal-imab-bg.org 2337 Fig. 1. PG of the tongue Fig.3. Recurring PG

Case 2: Fig. 4. Lobular capillary hemangioma A young male aged 17 without any comorbidities is referred to the Department of Oral surgery of FDM – Plovdiv because of suspicious formation, localized on the lingual frenulum (Fig. 2). The patient’s complaints of bleeding dur- ing eating date back 4 months ago, the formation mean- while increased its size. Clinical evaluation revealed 10-6 mm large, soft and painless formation localized on the lin- gual frenulum. Periodontal evaluation revealed plentiful deposits of plaque. A recurrence was discovered 1 month after surgical excision of the formation (Fig. 3). Healing process during the three month period after the second surgery was un- eventful. Histopathological diagnostics confirmed lobular capillary hemangioma (Fig. 4).

Fig. 2. PG on the lingual frenulum

Case 3: A 36-years old male without comorbidities is referred to the Department of Oral surgery because of esthetically disconcerting formation localized on the lower that some- times bleeds when traumatized (Fig. 5). The formation dates two months ago and the patient also reports having a do- mestic accident. Clinical evaluation reveals 7-5mm large formation that is soft, painless and with a tendency for pro- voked bleeding. Histopathological analysis after surgical excision revealed lobular capillary hemangioma.

2338 https://www.journal-imab-bg.org J of IMAB. 2019 Jan-Mar;25(1) Fig. 5. PG on lower lip Fig. 7. Non-lobular capillary hemangioma

Case 4: A 34-years old female in good general health with no Case 5: comorbidities, who gave birth two months ago, is referred A 33-years old male patient in good general health is to the Department of Oral surgery in FDM-Plovdiv with com- referred to the Department of Oral surgery because of 6- plaints of bleeding during brushing of teeth. Clinical 9mm sized formation in the region of teeth 33 and 34 (Fig. evaluation revealed multiple formations on the gingiva of 8). Patient’s complaints include bleeding in the mandibular from tooth 13 to tooth 23 and on the mandibular region when brushing the teeth, which started two months gingiva around tooth 43 (Fig. 6). Periodontal assessment ago. During clinical examination a soft-elastic formation established deposits of plaque and tartar. The gingiva had that bleeds upon mechanical provocation was discovered. pasty and edematous look and was painful with palpation. Periodontal status established plaque deposits and multiple Treatment included surgical excision and curettage deep in pathological pockets. Segment radiograph showed no evi- the underlying tissues. There is no evidence of recurrence dence of pathology in the underlying bone structures (Fig. two months later. Biopsy results showed non-lobular capil- 9). Excision with Er:Yag laser of the formation and laser lary hemangioma (Fig. 7). treatment of the adjacent soft tissues and the buccal cortical plate of tooth 34 was implemented (Fig. 10, Fig. 11 and Fig. Fig. 6. PG on the maxillary gingiva of female patient 12). Histopathological evaluation documented non-lobu- lar hemangioma. The patient received further instructions for personal oral hygiene. No recurrence was established in the 6th month follow-up.

Fig. 8. PG on gingiva of tooth 34

J of IMAB. 2019 Jan-Mar;25(1) https://www.journal-imab-bg.org 2339 Fig. 9. Radiograph of tooth 34. No pathological find- Fig. 12. Postoperative site ings in the bone

Case 6: Fig. 10. Parameters of Er:Yag laser A 39-years old female with no comorbidities com- plained of bleeding and discomfort during eating and brush- ing her teeth in the anterior palatal region. She gave birth four months prior to her visit in the Department of Oral sur- gery and she is currently lactating. Her complaints began two months ago. The patient also informed us about prior surgery in the region associated with removal of cyst at the anterior teeth 10 years ago. Clinical evaluation found gingival lesion between teeth 12 and 13 (Fig. 13). The pea- sized formation is soft and bleeds upon mechanic provoca- tion. Periodontal status revealed plaque and multiple peri- odontal pockets. Treatment included excision followed by deep curettage of the underlying tissues. Diagnosis upon biopsy was non-lobular capillary hemangioma. The follow- up after six months does not reveal any recurring lesions.

Fig. 13. PG on the palatal mucosa. Between teeth 22 Fig.11. Excision with Er:Yag laser and 23

2340 https://www.journal-imab-bg.org J of IMAB. 2019 Jan-Mar;25(1) DISCUSSION: Usually such lesions are clinically presented as sin- Bhaskar et al. report that PG makes up for 1,85% of gle nodulus or pedunculated papula with smooth or lobular all oral pathologies [2,10]. The term “PG” is somewhat in- surface, sometimes ulcerated, black color, various size from accurate because such formations is not associated with in- few mm to few cm. Matured lesions are characterized by fection, does not produce pus and there is no clinical or poorer vascularization, increased amount of collagen and histological evidence of true granulation tissue [3]. Instead, pink color. The formations are usually painless, but bleed mild trauma or inflammation leading to quick connective easily when provoked or spontaneously due to increased tissue overgrowth are associated with the majority of the vascularization. [2] Angelopoulos described them reported cases [6]. The infection may be caused by strepto- histologically as hemangiomatous granuloma, emphasiz- cocci and staphylococci bacteria [11]. ing the abundance of blood vessels and inflammatory na- The presence of background infection and - ture of the process [6]. There are two histological types of tous skin lesions is relatively uncommon. Most infectious PG: lobular capillary hemangioma and non-lobular capil- diseases are associated with exanthema-like reaction. A com- lary hemangioma [20]. The first type clinically presents as bination of angiomatous lesion and infection can be ob- sessile lesion in 66,4% of the cases and the second type – as served in cases with Caposi or Bartonella infec- pedunculated lesion in 77% of the cases. [6,16] Our experi- tion. [11] Lately angiopoetin and efrin B2 in other tumors ence with the sessile, lobular PG on the lingual frenulum of the blood vessels along with Bartonella hanselae, supports this differentiation. The lobular region of the lobu- B.Quintana and HPV8 are found in recurring cases [12]. PG lar PG has more blood vessels of small diameter compared is additionally influenced by viral oncogenes, , to the central region of non-lobular PG. The central region microscopic arterial malformations, as well as gene depres- of non-lobular type has increased amount of blood vessels sion in the fibroblasts. [13,14] that are surrounded by mesenchymal cells non-reactive for It was evident that has a role in the devel- a-smooth muscle actin and muscle-specific actin compared opment of PG in our patients. More than 5% of intraoral PG to the lobular region of lobular PG. [16] is observed in pregnancy, thus supporting the terms “preg- The diagnosis of PG can often be challenging be- nancy tumor” and granuloma gravidarum. [3] Such lesions cause of the similarities in the clinical presentation of many are frequent occurrences in pregnancy in the light of in- other intraoral angiomatous lesions. Differential diagnosis creased progesterone and estrogen levels. Hosseini et al. includes capillary hemangioma [21], periphery giant cell establish that gingival hypertrophies are seen during preg- granuloma, periphery ossifying fibroma, hyperplastic nancy and disappear in menopause. [15] Frequency of PG gingival inflammation, pregnancy tumor, Kaposi sarcoma, peaks in teenagers and young patients, notably females. metastatic carcinoma, , non-Hodgkin lym- [3,16] Our findings confirm its prevalence in females and phoma. teenagers. Surgical excision is treatment method of choice, fol- It was evident that pregnancy has a role in the devel- lowed by deep curettage of the lesion, combined with re- opment of PG in our patients. PG affects predominantly the moval of the periosteum and granulation tissue around the gingiva (in 75% of cases)[6] but can also occur on the lips, adjacent teeth. [2,9,] Recurrence is observed in 15,8% of tongue, oral mucosa and palate [2,7,8,14,17] which corre- the cases. [10] A lot of authors explore alternative treatment sponds to our clinical experience. approaches in order to prevent such recurrences, including Intraoral gingival forms are usually located in max- cryotherapy, laser excision, ethanol etc. [2] No recurrence 3 illa. [2,18] Extraoral presentations most often include the months following Er:Yag laser excision was found in our skin and seldom the GI tract. [2,4] Multiple PG develop Case 5. spontaneously or secondary to trauma, skin inflammation or immunosuppression, however such occasions are rela- CONCLUSION: tively rare. [1] The case of multiple PG we presented devel- PG is relatively uncommon finding in the oral cavity. oped on the background of poor oral hygiene and no pre- Its diagnostics are hindered by the similarity to other tu- dating trauma. A vicious cycle is at play because of pain morous and non-tumorous formations. The clinical experi- during brushing of teeth, which impairs the oral hygiene ence we documented demonstrates different clinical pres- even further. Our findings are in agreement with the conclu- entations and therapeutical approaches in order to avoid sions of other authors [19] and we support the notion that recurrences after surgical excision of the pyogenic one possible reason for this uncommon pathology can be granuloma. the impaired oral hygiene.

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Please cite this article as: Neychev DZ, Cholakova RB, Sbirkova TI, Kisselov SN, Bachurska SY, Atanasov DT. Pyogenic granulomas in the oral cavity: a series of cases. J of IMAB. 2019 Jan-Mar;25(1):2337-2342. DOI: https://doi.org/10.5272/jimab.2019251.2337

Received: 18/08/2018; Published online: 29/01/2019

Address for correspondence: Deyan Neychev Department of Oral Surgery, Faculty of Dental Medicine, Medical University - Plovdiv 3 Hr. Botev blvd., 4000 Plovdiv, Bulgaria E-mail: [email protected] 2342 https://www.journal-imab-bg.org J of IMAB. 2019 Jan-Mar;25(1)