Keywords
dark patches - lingual bumps - pigmented fungiform papillae - lingual fimbriae - normal
variant of oral mucosa
Introduction
The normal variant in the oral mucosa is defined as a normal anatomical structure,
with no pathological condition that can be found in the routine oral examination,
but it can be misdiagnosed as a pathological condition.[1] Median rhomboid glossitis,[2]
[3] erythema migrants,[4] hairy tongue,[1] fissured tongue,[5] prominence of circumvallate papilla,[1] lingual varices,[6] and ankyloglossia[7] are the common normal variants that can be found in the dorsum and ventral of the
tongue. Other variants such as pigmented fungiform papillae of the tongue and lingual
fimbriae, which are rare conditions, are not widely reported and explained in the
reference.
Pigmented fungiform papillae, which appear as dark patches localized in the papillae,
are a form of physiologic pigmentation. It needs to be explained and described in
more detail, as this condition may be confused with malignant pigmented lesions.[8] This condition is commonly observed among the dark-skinned people.[9]
Lingual fimbria, which also considered a normal variant, may be the result of alteration
of the growth of the tongue. It appears as small papules or bumps on the ventral surface
and at the side of the lingual frenulum. There are limited references that can explain
this condition, and it is sometimes confused with squamous papilloma or oral warts.[1]
This case reported dark patches and lingual bumps on the tongue, considered as pigmented
fungiform papillae and lingual fimbriae, in the young adult Javanese male and female.
This case report emphasizes the clinical features of combined pigmented fungiform
papillae and lingual fimbriae as a rare entity, not usually found in the routine intraoral
examinations. We presented this case to provide information to clinicians, in order
for them to be able to recognize this condition and conduct proper management, as
it is rare and may resemblance other pathology, rendering misdiagnosis and overtreatment.
Cases
Case 1
A 22-year-old Javanese man, with no systemic disease, came with several dark patches
over his tongue. Intraoral examination showed multiple asymptomatic macula, which
were brownish-black and shared clear border over the dorsolateral sinistra and dextral
of the tongue ([Fig. 1A]). Small dark patches on the lateral dextral of the tongue ([Fig. 1B]), and large dark patches on the lateral sinistra of the tongue ([Fig. 1C]). No abnormality is observed in other mucosa. The patient had this condition for
2 years, but there were no other symptoms associated with the patches such as pain,
numb, tingling, or burning sensation. The ventral surface of the tongue showed hairy-like
papules, laterally making a line to the lingual veins, which were red in color and
asymptomatic. Some of them appeared as minor “bumps” ([Fig. 1D]
[1E]).
Fig. 1 Multiple brownish-black patches over the dorsal (A), lateral dextral (B), lateral sinistra (C), and asymptomatic hairy-like or bumps on ventral surface of tongue (D, E).
The patient did not smoke, chew tobacco, or consume alcohol alcohol but was fond of
spicy foods. The physical examination indicated obesity with body mass index (BMI)
of 41.52. Within a year of routine oral health examination, no drugs or treatment
could remove the patches. The family history review showed that the dark patches over
the tongue did not appear in other family members. The condition on the dorsolateral
surface of the tongue can be identified as pigmented fungiform papillae and the ventral
surface of the tongue can be identified as lingua fimbriae.
Case 2
A 21-year-old Javanese woman came with dark patches over the tongue which had been
known for over 4 to 5 months. Dorsolateral examination of the tongue showed multiple
brownish-black, diffuse, and asymptomatic macula, ([Fig. 2A]). These patches appeared over the posterior and anterior dorsal surfaces of the
tongue; also, they came with dark brown prominent papilla on the lateral surface ([Fig. 2B]
[2C]). The ventral surface of the tongue showed multiple papules on the medial and lateral
lingual veins. These papules appeared light red in color with hair-like shape, but
some of them had a minor “bumps” shape, which mainly appeared beside the lingual frenulum
([Fig. 2D]
[2E]).
Fig. 2 Multiple brownish-black patches over the dorsal (A), lateral dextral (B), lateral sinistra (C), and asymptomatic multiple bumps on the ventral surface of the tongue (D, E).
The patient had routine oral examination along with orthodontic treatment, and she
had no complaints about the patches. Because of the asymptomatic condition, no treatment
or drugs were used to treat the tongue. The physical examination showed the normal
BMI was 24.78. The patient was prescribed meloxicam over the past 5 months for condyle
arthritis, and no systemic disease was diagnosed. The patient did not smoke, chew
tobacco, or consume alcohol but was fond of spicy foods. No abnormality was observed
in other mucosa. Other family members had no patches on the surface of their tongue.
The condition on the dorsolateral surface of the tongue can be identified as pigmented
fungiform papillae and the ventral surface of the tongue can be identified as lingua
fimbriae.
Management
In our cases, biopsy for histological examination and other supporting assessment
were not performed, because there were no complaints from the patients. So, we considered
them as normal variants of oral mucosa from the history, anamnesis, and clinical appearance.
There was no treatment conducted in both cases, because we found no complaints lodged
by these patients about their respective conditions. Information and education about
the patches and bumps were provided to the patients.
Follow-up
As there was no treatment conducted in these cases, outcomes were not observed, and
follow-ups were not scheduled. We advised the patients to visit a clinician if they
have complaints associated with the patches and the bumps on the tongue. Six months
later, the condition of the patches and bumps on the tongue turned out to be identical
to the first condition. No other conditions and clinical changes were observed in
case 1 ([Fig. 3A]
[D]) and case 2 ([Fig. 3E]
[F]).
Fig. 3 The follow-up 6 months later. The patches over the dorsal of the tongue (A), lateral dextral (B), lateral sinistra (C), and bumps on ventral of the tongue (D) (female patient). The patches over the dorsal (E) and bumps on ventral of the tongue (F) (male patient).
Discussion
Diagnosis of pigmented lesions of the oral mucosa and perioral is challenging. Generally,
the clinical aspects of oral mucosa pigmented lesions are sufficient in establishing
the diagnosis. However, in some cases, biopsy is necessary and, occasionally, for
histology and immunohistochemical stains such as melan-A and S-100 protein, it may
be required to prompt a correct diagnosis.[10] In cases of pigmented papillae fungiform, sometimes, biopsy is not needed. The diagnosis
can be based on typical clinical features. Most cases reported the pigmented papillae
fungiform diagnosis established on the basis of clinical features as presented in
[Table 1]. Most cases were found in women and affected the anterior and lateral borders of
the tongue. The clinical appearance of the macula [11]was multiple hyperpigmented papillae,, with round or polygonal pattern, and presenting
with diffuse margins.[9]
Table 1
The reported of pigmented papillae fungiform
|
No.
|
Gender
|
Ages
|
Races
|
Location of affected papillae
|
Clinical appearance
|
Reference
|
|
1
|
Women
|
40
|
Black
|
Anterior of the tongue
|
Multiple hyperpigmented papillae, with pattern rose petal appearance
|
[2]
|
|
2
|
Women
|
44
|
African
|
Anterior of the tongue
|
Multiple hyperpigmented papillae, with round or polygonal shape
|
[5]
|
|
3
|
Women
|
12
|
African
|
Anterior, lateral, and dorsum of the tongue
|
Multiple hyperpigmented papillae, presented as dark patches
|
[8]
|
|
4
|
Women
|
30
|
Black
|
Anterior of the tongue
|
Multiple hyperpigmented papillae, with pattern rose petal appearance
|
[9]
|
|
5
|
Women
|
13
|
Mexican
|
Anterior of the tongue
|
Multiple hyperpigmented papillae, with pattern rose petal appearance
|
[6]
|
|
6
|
Women
|
13
|
South Asian
|
Anterior of the tongue
|
Light to dark brown pigmentation, round or polygonal in shape, circumscribed to the
papillae, and resembling a cobblestone appearance
|
[7]
|
|
7
|
Women
|
12
|
Moroccan
|
Anterior of the tongue
|
Multiple hyperpigmented papillae, present in a diffuse and symmetrical pattern
|
[10]
|
|
8
|
Man
|
36
|
Indian
|
Anterior and lateral of the tongue
|
Multiple hyperpigmented papillae, present in a symmetric distribution
|
[11]
|
|
9
|
Women
|
25
|
Saudi
|
Anterior and lateral of the tongue
|
Brown macula with diffuse margins with multiple hyperpigmented papillae
|
[4]
|
|
10
|
Women
|
23
|
Not reported
|
Anterior and lateral of the tongue
|
Multiple hyperpigmented papillae, which are present in a diffuse pattern
|
[12]
|
|
11
|
Women
|
29
|
Not reported
|
Anterior, lateral, and dorsum of the tongue
|
Multiple hyperpigmented papillae, presented in a diffuse and symmetrical pattern
|
[13]
|
|
12
|
Man
|
11
|
Black Brazilian
|
Anterior and lateral of the tongue
|
Multiple hyperpigmented papillae
|
[14]
|
In most of the reports, pigmented papillae fungiform were found in dark-skinned children
or young adults.[12] Cases of pigmented papillae fungiform are still not widely reported in Asia. The
first pigmented papillae fungiform was reported in Asia by Scarf and Marks (2003),[13] and then by Tan et al ( 2014) among the Chinese.[14] In our cases, the pigmented papillae fungiform were found in a young Javanese man
and woman with white skin.
The pigmented papillae fungiform condition undergoes physiological pigmentation. The
dermoscopy shows several projections with hyperpigmented papillae resembling rose
petal pattern[15] or cobblestone pattern.[14]
[16] Rose petal pattern was found in 39.66%, and cobblestone pattern in 100%, of female
and male Chinese.[14] In our cases, both patients showed a cobblestone pattern. The pattern of pigmentation
in our cases was found in the dorsolateral surface of the tongue (Case 1) and involving
three to seven fungiform papillae in the lateral and anterolateral surfaces of the
tongue (Case 2). Based on classification, Case 1 is type 2 of pigmented papillae fungiform,
and Case 2 is type 1 of pigmented papillae fungiform. In some of the literature, the
classification of pigmentation consists of three types. Type 1 is characterized by
well-defined hyperpigmented of fungiform papillae on the anterolateral side and tip
of the tongue. Type 2 has hyperpigmentation involving three to seven few fungiform
papillae over the dorsal surface of the tongue. Type 3 is hyperpigmentation of all
fungiform papillae on the dorsum of the tongue.[11]
[17]
Histological examinations were not performed in our cases because there were no complaints.
This condition is also found in other cases, but histological examination of the case
of pigmented fungiform papillae was not performed. Robles-Méndez et al (2017) reported
histological examination revealed pigmentation in the basal layer of papillae and
melanophages in the lamina propria,[18] and in other cases showed slight lymphocytic infiltrate in the superficial.[19]
The general or systemic condition may have connection with pigmented papillae fungiform.
Karine Francine Docx et al (2016) mentioned that pigmented papillae fungiform can
be found in obese children with an earlier menarche (higher level of estrogen).[20] One of our cases also present in a young obese (higher BMI) individual, but the
other case presented normal BMI.
Our cases are unique that is, the pigmented papillae fungiform found accompany lingual
fimbriae. Lingual fimbriae are the papules, laterally making a line to the lingual
veins and lingual frenulum. Lingual fimbriae may be a result of incomplete apoptosis
of the ventral surface of the tongue. The tongue originates from the first, second,
and third pharyngeal arches and develops at the beginning of the fourth intrauterine
week.[21] During this phase, a deep sulcus develops in front of and on both sides of the tongue
which gives the tongue its mobility.[22] Normally, apoptosis separates the tongue from the floor of the mouth, and the only
tissue that remains to anchor the tongue to the mouth floor is the frenulum.[23] The incomplete apoptosis in the ventral surface of the tongue results a normal residual
tissue not completely reabsorbed by the body during the development and growth of
the tongue and which is called lingual fimbriae.[24]
There are few studies about lingual fimbriae, especially in pathogenesis and clinical
presentation. Both pigmented fungiform papillae lingual fimbriae of the tongue were
considered normal variants, with no positive correlation to each other in development
and incidence. Lingual fimbriae appear as the result of incomplete cell apoptosis,
while pigmented fungiform papillae are associated with pigmentation disorder. At present,
there are no findings that show interrelated causes in both conditions.
Pigmented fungiform papillae and lingual fimbria were considered normal variants,
and they were not malignant or any condition that is threatening. No specific treatment
is needed, but further investigation should be done by clinicians if there are progressive
changes or significant complaints from patients. Knowledge and ability to identify
those clinical appearances are key to the management of both cases because of the
rareness and the resemblance of the clinical features with more serious pathology,
rendering misdiagnosis and overtreatment. Further studies need to be done to investigate
if there is association between pigmented fungiform papillae and lingual fimbriae
of the tongue besides the clinical aspects.
