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Inflammatory fibrous hyperplasia: a rare case in a child

Hiperplasia fibrosa inflamatória: um caso raro em Odontopediatria

ABSTRACT

This paper aims to describe an inflammatory fibrous hyperplasia case caused by chronic irritation due to poor dental positioning after trauma. An 11-year-old female patient was sent to a dental specialty center with the complaint of a soft tissue growth in the place of an unerupted maxillary central incisor, causing her behavioral changes becoming more timid. After anamnesis and clinical examination it was noted that tooth 11 was in a vestibular position, covered with a hyperplastic lesion. Surgical treatment was performed and the histopathological exam revealed inflammatory fibrous hyperplasia. Over the course of the follow-up appointments, it was possible to observe improvement on the mentioned tooth and the patient’s satisfaction. In face of the low prevalence of inflammatory fibrous hyperplasia, it is worth mentioning that the tooth positioning, outside its correct alignment in the arch, can cause repeated trauma to the mucosa, and then turning into etiological factor to inflammatory fibrous hyperplasia, affecting even children and adolescents. Thus, the dentistry must be alert to establish a diagnosis and clinical treatment, in addition to monitoring these cases.

Indexing terms
hyperplasia; Oral pathology; Pediatric dentistry

RESUMO

O objetivo deste trabalho é apresentar um caso de hiperplasia fibrosa inflamatória provocada por irritação crônica decorrente do mau posicionamento dental após trauma. Uma adolescente, 11 anos, foi encaminhada ao Centro de Especialidades Odontológicas, com queixa de que o incisivo central superior não havia erupcionado e em seu lugar havia surgido uma lesão de tecido mole, acarretando mudanças no comportamento da mesma que se tornou mais tímida. Após anamnese e exame clínico, constatou-se a presença do dente 11 em posição bastante vestibularizada, coberto por lesão hiperplásica. Foi realizado o tratamento cirúrgico e o resultado do exame histopatológico revelou que se tratava de hiperplasia fibrosa inflamatória. Durante as consultas para acompanhamento, foi possível observar a melhora no posicionamento do dente em questão e a satisfação da adolescente. Diante da baixa prevalência de hiperplasia fibrosa inflamatória na população infantil, vale ressaltar que o posicionamento do dente, fora do seu alinhamento correto no arco, pode provocar traumas repetidos em mucosa e, com isso, tornar-se fator etiológico para hiperplasia fibrosa inflamatória, acometendo, inclusive, crianças e adolescentes. Assim, o cirurgião dentista deve estar atento para estabelecer um diagnóstico e tratamento clínico oportunos, além do acompanhamento destes casos.

Termos de indexação
Hiperplasia; Patologia bucal; Odontopediatria

INTRODUCTION

Fibrous hyperplasia is an inflammatory reaction of the connective tissue that is considered to be a benign soft-tissue tumor commonly found in the oral cavity due to irritation or chronic local trauma [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...

2 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...

3 Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.
-44 Palmeira ARBLS, Florêncio AG, Silva Filho JP, Silva UH, Araújo NS Non neoplastic proliferative lesions: a ten-year retrospective study. RGO, Rev Gaúch Odontol. 2013;61(4):543-7.]. This condition is often related to the use of poorly adapted dentures or other etiological factors, such as chronic irritation caused by diastemata, the sharp edges of teeth, treatment procedures (iatrogenic etiology), roots fragments or other trauma [33 Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.,44 Palmeira ARBLS, Florêncio AG, Silva Filho JP, Silva UH, Araújo NS Non neoplastic proliferative lesions: a ten-year retrospective study. RGO, Rev Gaúch Odontol. 2013;61(4):543-7.].

Inflammatory fibrous hyperplasia is rare in children and adolescents, ranging from 5,6% [55 Ataíde AP, Fonseca FP, Santos Silva AR, Jorge Júnior J, Lopes MA, Vargas PA. Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population. Otorhinolaryngol. 2016;90:241-244. http://dx.doi.org/10.1016/j.ijporl.2016.09.027.
https://doi.org/10.1016/j.ijporl.2016.09...
] to 8% [66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
] of specimens in the paediatric population. Thus, biopsies in the pediatric population are rare [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,55 Ataíde AP, Fonseca FP, Santos Silva AR, Jorge Júnior J, Lopes MA, Vargas PA. Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population. Otorhinolaryngol. 2016;90:241-244. http://dx.doi.org/10.1016/j.ijporl.2016.09.027.
https://doi.org/10.1016/j.ijporl.2016.09...
,66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
]. The treatment of choice is surgical excision and reoccurrence is uncommon [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,22 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...
]. A biopsy is important to confirm the diagnosis [66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
,77 Silva LVO, Arruda JAA, Martelli SJ, Kato CNAO, Nunes LFM, Vasconcelos ACU, et al. A multicenter study of biopsied oral and maxillofacial lesions in a Brazilian pediatric population. Braz Oral Res. 2018;32:e20. http://dx.doi.org/10.1590/1807-3107bor-2018
https://doi.org/10.1590/1807-3107bor-201...
] and it is essential for hyperplastic tumors, considering the clinical similarity of the different pathologies of this nature [88 Patankar SR, Tripathi NS, Gaonkar PP, Sridharan G. A rare case of giant cell fibroma in a pediatric patient. Clin Cancer Investig J. 2016;5:327-329.

9 Reddy V, Saxena S, Saxena S, Reddy M. Reactive hyperplastic lesions of the oral cavity: A ten year observational study on North Indian Population. J Clin Exp Dent. 2012; 4(3): 136-140. http://dx.doi.org/10.4317/jced.50670
https://doi.org/10.4317/jced.50670...
-1010 Reddy VK, Kumar N, Battepati P, Samyuktha L, Nanga SP. Giant cell fibroma in a paediatric patient: a rare case report case. Rep Dent. 2015;2015:240374. http://dx.doi.org/10.1155/2015/240374
https://doi.org/10.1155/2015/240374...
]. In the pediatric population, the most commonly studied conditions are related to dental caries [66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
,1111 Padovani MC, Santos MT, Sant’ Anna GR, Guaré RO. Prevalence of oral manifestations in soft tissues during early childhood in Brazilian children. Braz Oral Res. 2014;28:1-7. http://dx.doi.org/10.1590/1807-3107BOR-2014.vol28.0036
https://doi.org/10.1590/1807-3107BOR-201...
].

Few studies in the literature have specifically addressed inflammatory fibrous hyperplasia. Most publications describe this condition together with other reactive tumors [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,22 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...
] or associated with the use of poorly adapted dentures in adults [33 Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.]. This paper describes a rare case of inflammatory fibrous hyperplasia in a child caused by chronic irritation due to poor dental positioning.

CASE REPORT

An 11-year-old, dark-skinned female was sent to the Dental Specialty Center in Estância, a city in the state of Sergipe, Brazil, by a dentist from primary care, with complaint an unerupted maxillary central incisor. The clinical treatment started after the signature of the informed consent term.

During the anamnesis, good general health was determined and the permanent tooth remained in the early stage of eruption long after the primary incisor exfoliation. The patient reported to have dental trauma, resulting the permanent incisor getting covered with soft tissue. Resulting in change in patient behavior, became angry, introverted and ashamed to smile

The clinical exam revealed a round growth on the mucosa of the upper lip with a soft consistency, smooth surface, pediculate insertion as well as color and texture similar to the surrounding tissues. The maxillary right central incisor demonstrated ectopic eruption and was in a vestibular position, completely covered by the growth (figure 1, 2). The radiographic findings were not altered and compatible with the clinical examination.

Figure 1
Clinical aspect of soft tissue growth.
Figure 2
Clinical aspect of central incisor covered by the growth.

Treatment involved the surgical excision of the growth, vestibuloplasty and follow up to determine the correct tooth repositioning. The removed tissue was sent for histopathological analysis and the patient was released after receiving instructions regarding postoperative care.

The patient was evaluated one week after surgery and the suture was removed. There were neither reports of discomfort nor complications. The patient seemed satisfied with the treatment, which was confirmed by her mother. The histopathological analysis revealed inflammatory fibrous hyperplasia, with keratinized squamous epithelial tissue, dense fibrous underlying connective tissue and blood vessels congested with chronic inflammatory infiltrate (figure 3).

Figure 3
Histological aspect of growth.

The return visits at 4 and 12 months demonstrated improvements in the positioning of the tooth, which approached its physiological position without an orthodontic appliance assistance (figures 4-6).

Figure 4
mmediate postoperative phase.
Figure 5
Four-month follow up.
Figure 6
12-month follow up

DISCUSSION

Inflammatory fibrous hyperplasia is rare in children and adolescents and the literature reports a greater incidence in this population between the ages of 9 and 14 [66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
,99 Reddy V, Saxena S, Saxena S, Reddy M. Reactive hyperplastic lesions of the oral cavity: A ten year observational study on North Indian Population. J Clin Exp Dent. 2012; 4(3): 136-140. http://dx.doi.org/10.4317/jced.50670
https://doi.org/10.4317/jced.50670...
], which is in agreement with the case reported herein. This condition is the result of chronic low-intensity trauma, which is most often caused by poorly adapted dentures [33 Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.,44 Palmeira ARBLS, Florêncio AG, Silva Filho JP, Silva UH, Araújo NS Non neoplastic proliferative lesions: a ten-year retrospective study. RGO, Rev Gaúch Odontol. 2013;61(4):543-7.]. In the present case, however, chronic irritation resulting from the maxillary central incisor poor positioning, was the probable etiological agent.

The hyperplastic growth led to behavioral changes in the adolescent, such as irritability as well as social contact avoidance and smile embarrassment, which interfering negatively in quality of life. Similar behavior is reported studies addressing the impact of oral-dental abnormalities on adolescents’ quality of life [1212 Benson PE, Da’as T, Johal A, Mandall NA, Williams AC, Baker SR. et al. Relationships between dental appearance, self-esteem, socio-economic status, and oral health-related quality of life in UK schoolchildren: A 3-year cohort study. Eur J Orthod. 2015;37(5):481-90. http://dx.doi.org/10.1093/ejo/cju076
https://doi.org/10.1093/ejo/cju076...
]. Such changes were gradually reversed following the surgical removal of the growth and the return of the tooth to its physiological position.

This condition could have been minimized by a timely professional assessment and adequate case follow up, which did not occur due to a lack of information from the part of the patient’s guardian as well as the absence of immediate signs and symptoms. According to Reddy et al. [1010 Reddy VK, Kumar N, Battepati P, Samyuktha L, Nanga SP. Giant cell fibroma in a paediatric patient: a rare case report case. Rep Dent. 2015;2015:240374. http://dx.doi.org/10.1155/2015/240374
https://doi.org/10.1155/2015/240374...
], the search for professional care only occurs when a lesion produces symptoms or compromises oral esthetics.

A biopsy is of considerable importance to a correct diagnosis [66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
,77 Silva LVO, Arruda JAA, Martelli SJ, Kato CNAO, Nunes LFM, Vasconcelos ACU, et al. A multicenter study of biopsied oral and maxillofacial lesions in a Brazilian pediatric population. Braz Oral Res. 2018;32:e20. http://dx.doi.org/10.1590/1807-3107bor-2018
https://doi.org/10.1590/1807-3107bor-201...
]. However, the diagnosis in cases such as this one constitutes a challenge due to the similar clinical characteristics found among hyperplastic growths. For instance, giant cell granuloma and pyogenic granuloma are part of the differential diagnosis with inflammatory fibrous hyperplasia [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,44 Palmeira ARBLS, Florêncio AG, Silva Filho JP, Silva UH, Araújo NS Non neoplastic proliferative lesions: a ten-year retrospective study. RGO, Rev Gaúch Odontol. 2013;61(4):543-7.,88 Patankar SR, Tripathi NS, Gaonkar PP, Sridharan G. A rare case of giant cell fibroma in a pediatric patient. Clin Cancer Investig J. 2016;5:327-329.

9 Reddy V, Saxena S, Saxena S, Reddy M. Reactive hyperplastic lesions of the oral cavity: A ten year observational study on North Indian Population. J Clin Exp Dent. 2012; 4(3): 136-140. http://dx.doi.org/10.4317/jced.50670
https://doi.org/10.4317/jced.50670...

10 Reddy VK, Kumar N, Battepati P, Samyuktha L, Nanga SP. Giant cell fibroma in a paediatric patient: a rare case report case. Rep Dent. 2015;2015:240374. http://dx.doi.org/10.1155/2015/240374
https://doi.org/10.1155/2015/240374...
-1111 Padovani MC, Santos MT, Sant’ Anna GR, Guaré RO. Prevalence of oral manifestations in soft tissues during early childhood in Brazilian children. Braz Oral Res. 2014;28:1-7. http://dx.doi.org/10.1590/1807-3107BOR-2014.vol28.0036
https://doi.org/10.1590/1807-3107BOR-201...
]. Despite the importance of a biopsy, between 7,4% and 17% of specimens were from a paediatric population [55 Ataíde AP, Fonseca FP, Santos Silva AR, Jorge Júnior J, Lopes MA, Vargas PA. Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population. Otorhinolaryngol. 2016;90:241-244. http://dx.doi.org/10.1016/j.ijporl.2016.09.027.
https://doi.org/10.1016/j.ijporl.2016.09...
,66 Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
https://doi.org/10.1111/adj.12174....
]. The present case demonstrates the value of the anatomopathological examination, which was performed in the public health system.

The literature proposes excision as the treatment of choice in cases of inflammatory fibrous hyperplasia, with the complete removal of the growth, along with a safety margin [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,22 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...
]. This therapeutic approach was adopted in the present case and was combined with vestibuloplasty to establish the depth of the vestibule. The tissue removed was sent for histopathological analysis and the findings were compatible with data described in the literature for the inflammatory fibrous hyperplasia diagnosis [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...

2 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...
-33 Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.]. Researchers consider the prognosis excellent provided that the etiological agent is removed [11 de Santana Santos T, Martins-Filho PR, Piva MR, de Souza Andrade ES. Focal fibrous hyperplasia: a review of 193 cases. J Oral Maxillofac Pathol. 2014; 18(1): 86-89. http://dx.doi.org/10.4103/0973-029X.141328
https://doi.org/10.4103/0973-029X.141328...
,22 Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
https://doi.org/10.1155/2012/370242...
], as demonstrated in the present case, in which no recurrence was observed during the 12 months of follow up.

CONCLUSION

The tooth position out of its correct alignment in the arch can cause repeated trauma to the mucosa, resulting in inflammatory fibrous hyperplasia, even in the young population.

REFERENCES

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    » https://doi.org/10.4103/0973-029X.141328
  • 2
    Vergotine RJ. A giant cell fibroma and focal fibrous hyperplasia in a young child: a case report. Case Rep Dent. 2012;2012:370242. http://dx.doi.org/10.1155/2012/370242
    » https://doi.org/10.1155/2012/370242
  • 3
    Barros RMG, Campos KSM, Cabral LM. Relato de caso clínico de hiperplasia fibrosa inflamatória. Rev Odontol Araçatuba. 2014;35(2):15-18.
  • 4
    Palmeira ARBLS, Florêncio AG, Silva Filho JP, Silva UH, Araújo NS Non neoplastic proliferative lesions: a ten-year retrospective study. RGO, Rev Gaúch Odontol. 2013;61(4):543-7.
  • 5
    Ataíde AP, Fonseca FP, Santos Silva AR, Jorge Júnior J, Lopes MA, Vargas PA. Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population. Otorhinolaryngol. 2016;90:241-244. http://dx.doi.org/10.1016/j.ijporl.2016.09.027.
    » https://doi.org/10.1016/j.ijporl.2016.09.027.
  • 6
    Ha WN, Kelloway E, Dost F, Farah CS. A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population. Aust Dent J. 2014;59(2):221-225. http://dx.doi.org/10.1111/adj.12174.
    » https://doi.org/10.1111/adj.12174.
  • 7
    Silva LVO, Arruda JAA, Martelli SJ, Kato CNAO, Nunes LFM, Vasconcelos ACU, et al. A multicenter study of biopsied oral and maxillofacial lesions in a Brazilian pediatric population. Braz Oral Res. 2018;32:e20. http://dx.doi.org/10.1590/1807-3107bor-2018
    » https://doi.org/10.1590/1807-3107bor-2018
  • 8
    Patankar SR, Tripathi NS, Gaonkar PP, Sridharan G. A rare case of giant cell fibroma in a pediatric patient. Clin Cancer Investig J. 2016;5:327-329.
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    Reddy V, Saxena S, Saxena S, Reddy M. Reactive hyperplastic lesions of the oral cavity: A ten year observational study on North Indian Population. J Clin Exp Dent. 2012; 4(3): 136-140. http://dx.doi.org/10.4317/jced.50670
    » https://doi.org/10.4317/jced.50670
  • 10
    Reddy VK, Kumar N, Battepati P, Samyuktha L, Nanga SP. Giant cell fibroma in a paediatric patient: a rare case report case. Rep Dent. 2015;2015:240374. http://dx.doi.org/10.1155/2015/240374
    » https://doi.org/10.1155/2015/240374
  • 11
    Padovani MC, Santos MT, Sant’ Anna GR, Guaré RO. Prevalence of oral manifestations in soft tissues during early childhood in Brazilian children. Braz Oral Res. 2014;28:1-7. http://dx.doi.org/10.1590/1807-3107BOR-2014.vol28.0036
    » https://doi.org/10.1590/1807-3107BOR-2014.vol28.0036
  • 12
    Benson PE, Da’as T, Johal A, Mandall NA, Williams AC, Baker SR. et al. Relationships between dental appearance, self-esteem, socio-economic status, and oral health-related quality of life in UK schoolchildren: A 3-year cohort study. Eur J Orthod. 2015;37(5):481-90. http://dx.doi.org/10.1093/ejo/cju076
    » https://doi.org/10.1093/ejo/cju076

Publication Dates

  • Publication in this collection
    25 Apr 2019
  • Date of issue
    2019

History

  • Received
    12 Mar 2018
  • Reviewed
    08 June 2018
  • Accepted
    30 Oct 2018
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