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 Table of Contents  
REVIEW ARTICLE
Year : 2019  |  Volume : 8  |  Issue : 1  |  Page : 1-4

Nostalgia of neonatal Bednar's aphthae


1 Department of Oral Medicine and Radiology, Faculty of Dental Sciences, SGT University, Gurgaon, Haryana, India
2 Department of Oral and Maxillofacial Pathology, Faculty of Dental Sciences, SGT University, Gurgaon, Haryana, India

Date of Web Publication29-Jan-2019

Correspondence Address:
Dr. Astha Chaudhry
Flat No 102, Tower G, Corona Optus, Sector 37C, Gurgaon - 122 001, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_71_18

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  Abstract 


Aphthous ulcers have been extensively studied and researched throughout the English literature. Its various types and mimicking lesions have been very well reviewed. However, ulcers of neonates also called as Bednar's aphthae have not been well studied and documented. It was initially mentioned in the pediatric literature in the 1900s and was thought to be rare. Recent studies suggest that it is not as uncommon as is supposed to be but is frequently undiagnosed. The purpose of this review is to bring this common, yet uncommonly reported anomaly to the readers, especially oral physicians and pediatricians so as to have an insight into this neonatal pathosis for the effective diagnosis and management.

Keywords: Aphthous, neonate, palate, ulcer


How to cite this article:
Chaudhry A, Saluja P. Nostalgia of neonatal Bednar's aphthae. J Clin Neonatol 2019;8:1-4

How to cite this URL:
Chaudhry A, Saluja P. Nostalgia of neonatal Bednar's aphthae. J Clin Neonatol [serial online] 2019 [cited 2019 Apr 23];8:1-4. Available from: http://www.jcnonweb.com/text.asp?2019/8/1/1/250982




  Introduction Top


An ulcer is defined as a breach in the continuity of the oral epithelium associated with molecular degeneration. An aphtha is a term commonly used to describe small ulceration in the mouth.

Recurrent aphthous ulcers/stomatitis (RAS), also known as canker sore or stress ulcers, is one of the most common oral pathoses. Oral medicine specialists and investigators no longer consider RAS to be a single disease but, rather, several pathologic states with similar clinical manifestations.[1] Those lesions have also been called by other authors as aphthous-like lesions. The list includes Behcet's syndrome, celiac disease, cyclic neutropenia, nutritional deficiencies, immunoglobulin A deficiency, and immunocompromised conditions, including HIV disease, inflammatory bowel disease, mouth and genital ulcers with inflamed cartilage syndrome, periodic fever, aphthous stomatitis, pharyngitis and cervical adenitis syndrome, Reiter's disease, Sweet's syndrome, and ulcus vulvae acutum.[2] Allergic or psychological abnormalities have also been implicated in the cases of RAS.

All these lesions show similar types of ulcers clinically, which can be classified as minor, major, or herpetiform. These differences are essentially clinical and correspond to the degree of severity. The ulcers are typically painful, oval, symmetric, and shallow covered by a yellow fibrinous membrane and surrounded by an erythematous halo.

Bednar's aphthae is a class of ulcers that is not related to the conventional group of RAS but has a similar clinical appearance. It owes its name to the pediatrician named Alois Bednar, who described the same in 1850.[3] To avoid confusion with the conventional terminology of an aphthous group of lesions, Epstein et al. referred it to be “ulceration of the palatine angles.” However, it is still referred to as Bednar's aphthae throughout the literature.

It was described in the ancient pediatric literature in the 19th century.[4],[5] It got extinct from the literature as the PubMed search of the English literature showed only four reports using the keywords Bednar aphthae, neonatal aphthae, and ulcera pterygoidea. In a prospective study by Nebgen et al., it was found that these ulcers were present in 15.8% of healthy neonates, suggesting that it is not a rare phenomenon. Hence, the main aim is to bring this forgotten disease in the existing knowledge of the readers.


  Clinical Features Top


Bednar's aphthae also called as “ulcera pterygoidea” are described as small palatal ulcers typically seen in otherwise healthy newborn infants. These ulcers can be seen in infants of 2 days of age up to 6 weeks. The typical location is at the junction of the hard and soft palate. They are usually described as bilaterally symmetrical located medial to the faucial pillars.[3] Like aphthae, they are shallow circumscribed ulcers about the size of pea or a bean with central exudation surrounded by a hyperemic halo [Figure 1]a. They may be covered by a yellowish-white fibrinous layer, which is firmly adherent to the base and may bleed on removal. They may be extremely painful when the infant sucks. Due to the compromised nutrition, the infant may be highly irritable and may display inconsolable crying during feeding. They may be preceded by hyperemia of the mucosa as seen in aphthous ulcers.
Figure 1: (a) Illustration showing the appearance of Bednar's aphthae. (b) Bednar's aphthae along with mid palatine raphe ulcer. (c) Confluence of the two with resultant butterfly shape

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Nebgen et al.[3] on examining 1654 infants categorized the ulcers into small solitary lesions and larger confluent ones. The smaller ones can be unilateral or bilateral, whereas the confluent ones were always bilateral. Occasionally, the bilateral ulcers may be joined by a bridge. In some cases along with bilateral ulcers, ulcer on the midpalatal raphe may develop which may confluence together to produce the shape of the butterfly with its body formed by the central ulcer and wings represented by the symmetric lateral ulcers [Figure 1]b and [Figure 1]c. These have been misnamed as “pseudodiphtheria” although they have no resemblance to the clinical picture of diphtheria.

Since the lymph and blood vessels are opened by the ulcerative process, these ulcers may be the cause of sepsis in the newborn.[6],[7],[8],[9],[10]

The reduced number of reports is due to the fact that the oral cavity of the newborn infant is seldom examined with a tongue depressor and a routine oropharyngeal aspiration is rarely performed to avoid any trauma to the fragile mucosa.

They are self-limiting and have been reported to heal within 1–4 weeks.


  Etiopathogenesis Top


The etiology is obscure. Multiple hypotheses have been proposed. In the 1900s, it was believed to be traumatic in origin caused by the way the newborn's mouth is cleaned directly after birth or following meals[9] by the use of cloth for wiping the oral cavity[11] or through a mechanical trauma by a hard teat or pacifier.[12] Why this act of cleaning caused ulcers in this particular location is due to the fact that the mucosa of the palate is tensely stretched and can become the seat of superficial lesions during nursing or washing the mouth. Some authors also suggested that these should be included in the category of stomatitis decubitalis, as the border of transition of the hard palate has sharp wing-like protrusions, which on application of pressure during wiping may lead to erosion of the mucosa and subsequent ulcerations similar to decubitus ulcers (bedsores).

Later, it was thought to be due to the traumatic nature of the nipple of the feeding bottle and due to the horizontal position of feeding. As reported by Tricarico et al.[13] in a case, bilateral ulcers subsided on changing the nipple and feeding position. However, the occurrence of bilaterally symmetrical lesions as classically described by Bednar through a traumatic origin is questionable.

Nebgen et al.[3] mentioned that these ulcers are seen in infants born spontaneously stating that vaginal delivery exposes the neonates with the vaginal microbiota, thus suggesting an infectious origin for these ulcers. Furthermore, these ulcers were not seen in preterm infants, but the association with these is not well explained.

An interesting observation was made by Nebgen et al.[3] that these lesions were seen in infants on nutritional formula. Infant formula is made as similar to breast milk as possible, but there are multiple reports on the risk of development of immunologically mediated diseases such as type I diabetes in infants dependent on formula feeding.[14] This is explained due to the exposure and increased the permeability of the neonatal gut to the dietary antigens, especially cow's milk proteins which triggers an autoimmune response inducing a pro-inflammatory state and inhibiting the suppression of cytotoxic T-lymphocytes, which eventually destroy pancreatic islet beta-cells. Maternal milk contains host defense factors such as antibacterial peptides, lysozyme, and secretory immunoglobulins not found in the formula, which protects the child against these disorders.[15]

There are increased reports of development of allergy and asthma in infants on formula feeding.[16] The reason is due to the development of IgE-mediated responses on the early exposure to the foreign antigens in the formula. It is known that environmental factors affect gene expression and manifestation of disease. Early fetal exposures to nutrition and other environmental factors may program organ development and the future development of disease.

Bednar's aphthae was hypothesized to be the result of nonspecific immunological reaction to the antigens; a neonate is exposed to in the early days of life. This hypothesis has a factual base in that these ulcers are mainly localized in the tonsillar region. The tonsillar region in constitution with the pharyngeal immune compartment represents a specialized mucosa-associated lymphoid tissue consisting of the buccal mucosa, salivary glands, and Waldeyer's ring. These act as inductive sites where most lymphocytes are activated and expanded on antigen stimulation and constitute the innate immune response of the oral cavity.[17]

In the case reported by Tricarico et al., antibodies such as antinuclear antibody, extractable nuclear antigens, antismooth muscle antibody, and antibody against Sjogren syndrome were tested positive in the child suggesting some immunological pathology. However, they were also positive in the mother suggesting a possible vertical transmission. Antinuclear antibodies (ANA) are autoantibodies that bind to contents of the cell nucleus and are mainly found in autoimmune disorders, infections, and in 5% of healthy individuals also.[18] These antibodies help in monitoring the progression of disease. In the reported case also, the level of ANA decreased after 4 months.

These hypotheses are very interesting, but no studies have been done in this regard so far. The practical difficulties include the reduced and difficult examination of the oral cavity of the newborn and difficulty in endoscopic examinations and biopsies.


  Management Top


Since etiology is not well defined, so is the management. Some authors advocate that these ulcers are not very harmful and do not cause much problems except difficulty during feeding. Hence, no specific treatment is advocated since they are known to regress spontaneously from a period between 1 and 4 weeks.

Considering the traumatic etiology, various modifications have been proposed. The use of breastfeeding, orthodontic nipples, change in the feeding position, and increase in the hole size of nipple have been advocated.

Breastfeeding is the best possible alternative as there will not be a question of traumatic action of nipple nor immunological pathology by the formula. Orthodontic nipples are asymmetrically shaped and designed to mimic the natural breast, which gets flattened in baby's mouth when baby feeds. They fit the shape of baby's palate and gums, giving the tongue and jaw more room to move naturally while sucking, and promoting healthy oral development.

The horizontal feeding position has also been implicated in the development of soft palatal ulcers.[19] The slightly upright position during feeding resulted in the resolution of ulcers in the study by Nebgen et al.[3] and in the report by Tricarico et al.[13]

The use of smaller hole size of the nipple may also cause increased pressure during sucking and may have a role in causation, and changes in hole diameter of the nipple have also shown positive results in the study.

The symptomatic relief can be provided by topical anesthetics or glycerine with the use of earbud, but it is difficult to apply. The glycerine will act as coating agent and provide relief during feeding. The historic reports in the literature have suggested the use of 2% silver nitrate.

Due to the ulcerative process, the lymph and blood vessels are opened by this process and this may act as source of sepsis in the newborn, especially in a debilitated infant. Hence, care has to be taken for the infant to prevent the complications.


  Conclusion Top


Aphthous ulcers are well known to both pediatricians as well as oral physicians. However, these bilateral palatine ulcers of neonates and infants misnamed as Bednar's aphthae are less known even though they are not uncommon. Hence, such commonly prevalent yet poorly studied pathologic entity should be known by pediatricians, neonatologists, as well as the oral physicians, so that the necessary studies regarding the etiopathogenesis and treatment can be carried out to better understand this pathology. The thorough knowledge about their clinical presentation is also required to avoid useless examinations and invasive diagnostic tests and also to reduce the parental anxiety by educating them about the self-limiting nature of this oral pathosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Greenberg M, Glick M, Ship J. Burkets Oral Medicine. 11th ed. USA: PMPH; 2008.  Back to cited text no. 1
    
2.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2nd ed. USA: W.B. Saunders; 2002.  Back to cited text no. 2
    
3.
Nebgen S, Kasper HU, Schäfer D, Christ H, Roth B. Bednar's aphthae in neonates: Incidence and associated factors. Neonatology 2010;98:208-11.  Back to cited text no. 3
    
4.
Khitrov VI. Diagnosis of chronic erosion of palatal mucosa (Bednar's aphthae) in infants. Pediatriia 1976;6:67-8.  Back to cited text no. 4
    
5.
Adrion W, Albrecht W, Blumenfeld F, Finder G, Harmer L, Hinsberg V, et al. The Diseases of the Airways and the Oral Cavity: Third Part Acute and Chronic Odoritis, Pharynx, Larynx, Air - Tube and Bronchial Diseases · Abnormalities of Foreign Objects · Distortions. 1st ed. Berlin: Springer; 1928.  Back to cited text no. 5
    
6.
Bohn H. The Childrens' Oral Diseases. Leipzig: Verlag Wilhelm Engelmann; 1866.  Back to cited text no. 6
    
7.
Brecelj A. Uber einen pseudodiphtherischen symptomkomplex bei Neugeborenen. Jahrb Kinderheilkd 1904;59:54-73.  Back to cited text no. 7
    
8.
Epstein A. Uber septische erkrankungen der schleimhaute bei kindern, Arch Kinderheilkd 1880;1:25-45.  Back to cited text no. 8
    
9.
Epstein A. Uber pseudodiphtheritis septhamischen ursprungs bei Neugeborenen und Sauglingen. Jahrb Kinderheilkd 1895;39:420-40.  Back to cited text no. 9
    
10.
Linzenmeier G. Sepsis bei neugeborenen, ausgehend von den Bednar'schen aphthen. Zentralbl Gynakol 1911;50:S35-7.  Back to cited text no. 10
    
11.
von Berlin-Heimandahl S. Die Krankheiten des Neugeborenen und Fruhgeborenen. Stuttgart: Enke; 1960.  Back to cited text no. 11
    
12.
Ewerbeck H. Infant Physiology, pathology and therapy in the first year of life. Heidelberg: Springer; 1962.  Back to cited text no. 12
    
13.
Tricarico A, Molteni G, Mattioli F, Guerra A, Mordini B, Presutti L, et al. Nipple trauma in infants? Bednar aphthae. Am J Otolaryngol 2012;33:756-7.  Back to cited text no. 13
    
14.
Patelarou E, Girvalaki C, Brokalaki H, Patelarou A, Androulaki Z, Vardavas C, et al. Current evidence on the associations of breastfeeding, infant formula, and cow's milk introduction with type 1 diabetes mellitus: A systematic review. Nutr Rev 2012;70:509-19.  Back to cited text no. 14
    
15.
Caicedo RA, Li N, Des Robert C, Scumpia PO, Hubsher CP, Wasserfall CH, et al. Neonatal formula feeding leads to immunological alterations in an animal model of type 1 diabetes. Pediatr Res 2008;63:303-7.  Back to cited text no. 15
    
16.
Alison S. The risks of not breastfeeding for mothers and infants. Rev Obstet Gynecol 2009;2:222-31.  Back to cited text no. 16
    
17.
Wu RQ, Zhang DF, Tu E, Chen QM, Chen W. The mucosal immune system in the oral cavity-an orchestra of T cell diversity. Int J Oral Sci 2014;6:125-32.  Back to cited text no. 17
    
18.
Tan EM, Feltkamp TE, Smolen JS, Butcher B, Dawkins R, Fritzler MJ, et al. Range of antinuclear antibodies in “healthy” individuals. Arthritis Rheum 1997;40:1601-11.  Back to cited text no. 18
    
19.
Pedra C, Terra CM, Ejzenberg B, Baldacci ER, Okay Y. Oral palatine ulcers of a traumatic nature in infants: Bednar's aphthae. Int J Pediatr Otorhinolaryngol 1996;35:39-49.  Back to cited text no. 19
    


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Abstract
Introduction
Clinical Features
Etiopathogenesis
Management
Conclusion
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