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Cheilitis Granulomatosa

MED ARH 2011; 65(6): 373-374

doi: 10.5455/medarh.2011.65.373-374

Case report

Cheilitis Granulomatosa
Mirjana Gojkov-Vukelic, Sanja Hadzic, Alma Kantardzic
Department for Oral Medicine and Periodontology, Faculty of Dentistry, Sarajevo, Bosnia and Herzegovina

heilitis granulomatosa is a rare disease characterised by the recurrent


labial swelling of one or both lips with the possibility of the condition
to remain on a permanent basis. The disease may appear independently
or it may be linked to a paralysis such as the facial and lingua plicata which
then characteristic of the Melcersson-Rosenthal Syndrome. The aim of this
paper is to show a case of a patient with the granulomatosae cheilitis and
lingua plicata whose reaction to the Chymoral Forte treatment was excellent.
Key words: Macrocheilia, Cheilitis granulomatosa, Melcersson
Rosenthal Syndrome

Corresponding author: Mirjana Gojkov-Vukelic, MDD, Department for Oral Medicine and Periodontology,
Faculty of Dentistry, Sarajevo, Bosnia and Herzegovina. E-mail: gojkov@bih.net.ba

1. Introduction
The orofacial granulomatosis is described as the uncommon granulomatosae condition that leads to a painless
symmetric edema of the orofacial tissues. There are two clinical forms: Cheilitis granulomatosa, and the Melcersson-Rosenthal Syndrome. The possible
causes are localized form of sarcoidosis, the atypical allergy, manifestation
of enteritis (Crohns disease), dental focal inflammation and others.
The granulomatosae cheilitis is clinically characterized by the diffuse increase of the lips (one or both), rarely
followed by the swelling of the gingiva,
the buccal mucosa and the palate (1).
The disease can appear independently
or it may be linked to a paralysis such as
the facial and lingua plicata which then
characteristic of the Melcersson-Rosenthal Syndrome (2). The provisional diagnosis is established with the exclusion of other granulomatosae developments where it is possible to identify
the causes of their occurrence. The incisional biopsy with PHD confirms or
excludes the granulomatose character
of the development and the established
diagnosis of Cheilitis granulomatosa.

The histological characteristic of


the disease is the uncommon lip granuloma. On the grounds of recent research into atypical cases, it was concluded that the lymphatic inflammatory
infiltrates occur in the submucous layers of tissue together with the uncommon granuloma linking the illness to
tissue edema (3, 4).
The differential diagnosis of cheilitis granulomatosa is extensive and includes amyloidosis, cheilitis glandularis, sarcoidosis, Crohns disease, cheilitis actinica, neoplasma and infection
such as tuberculosis and syphilis (5).
The aim of this paper is to show a
case of a patient with the granulomatosae cheilitis and lingua plicata whose
reaction to the Chymoral Forte treatment was excellent.

2. Case Overview
A 61-year old male was admitted to
the Clinic for Oral Medicine and Periodontology in October 2008 due to a
diffuse painless edema of the upper lip.
His medical history indicates that
the swelling of the lip had occurred earlier and that it was more prominent on
the upper lip but never in the current

proportions which the led the patient


to have it examined. The patient notes
that he has had high blood pressure
for the last two years. Since then, he
has been subjected to an antihypertensive therapy (Lopril H) and an aspirin
on daily basis for preventive purposes,
while answering negative to any other
illnesses. He does not link the swelling
of the lip to the season, the food which
he has consumed nor allergy. The clinical examination showed that the lip
was of solid consistency, light pink colour, smooth and shiny. Upon palpation,
granulomatosis developments the size
of a large pea are felt (Figure 1).

Figure 1. Granulomatosis developments the size


of a large pea are felt.

Other mucosa did not have pathological changes, lingua plicata present.
After conducting an examination
of the teeth and the periodontium, a
chronic periodontitis was diagnosed.
The OPG image analysis verified the
presence of the horizontal and vertical
absorption of the alveolar bone (Figure
2). No periapical developments on the
roots of the front teeth were established
thus excluding the dentogenic cause of
the lip swelling.
We recommended the patient to undergo incisional biopsy of the lip in or-

MED ARH 2011; 65(6): 373-374 Case report

373

Cheilitis Granulomatosa

without incisional biopsy and PHD.

Figure 2. Orthopantomogram of our patient

der to establish an accurate diagnosis


which the patient refused.
The lab blood and urine tests (the
complete blood count, sedimentation,
glucoses, thyroid gland hormones)
all registered normal values. The
alimentary and inhalation prick tests
were negative. The examination of the
stool for parasites and fungi was also
negative.
We recommended treatment with
Chimoral forte drag and claddings with
1% boric acid for 15 days. At the control check-up after five days, the swelling was significantly smaller and the
patient subjectively claimed to feeling
better initiating the continuation of the
therapy. After ten days, the lip swelling
was not present anymore (Figure 3).

Figure 3. The lip swelling was not present after


ten days

Despite the fact that the granulomatosae cheilitis has recently been described as part of the clinical symptoms
in the initial stage of the Crohns disease, the lab tests which we performed
could not confirm this (6). The presence of and linguae plicate indicates to
a monomorphic form of MelcerssonRosenthal Syndrome - Cheilitis granulomatosae. Despite performing detailed
clinical and lab testing, we could not
confirm the above mentioned diagnosis
374

3. Discussion
The diagnosis of a Cheilitis
granulomatosae is basically clinical and is verified by means of a biopsy. The blood tests might indicate
to increased levels of sedimentation,
lymphocytosis, peripheral eosinophilia, and in some cases increased
levels of IgA.
Numerous medicaments have
been used to treat labial edema:
systematic antibiotics, salazosulfapyridine, radio therapy and systematic, intralesional, topical corticosteroids. Some authors have used medicine against leprosy such as clofazimine (Lampren) in patients with cheilitis granulomatosa and have managed
to reduce the labial edema (1). In their
paper, Camacko Alonso et al. presented
the application of the systematic and intralesional corticosteroids and clofazimine which had produced satisfactory
results in all cases (7). Va-Galvn et
al. applied the intralesional corticosteroid (triamcinolone acetonide 10 mg /
mL) and achieved plausible improvements after three months. The patient
is currently subjected to maintenance
therapy with triamcinolone injections
twice a month and there was no recidivism during the monitored two-year
period (8). Other authors have applied
the intralesional corticosteroid (triamcinolone 20mg/ml, 1ml every 15 days
during a period of 60 days) and clofazimine (50 mg/day) for 90 days. The
complete relinquishment of the swelling was registered of 4 weeks of corticosteroid therapy (9). Pigozzi and al.
performed successful treatment of a
31-year old woman with MelcerssonRosenthal Syndrome using lymecycline
after an unsuccessful therapy with corticosteroids independently or in combination with the antihistamines, sulfasalazine and clofazimine (10). In our
case, by using Chymoral Forte (3x2
drag) we achieved complete remission
of the labial edema within the first five
days of the treatment. Chymoral Forte
has an anti-inflammatory, antiexudative and mucolytic action. During the
inflammatory process, the circulation
of blood and lymph is hindered due to
the sedimentation of fibrin and macro-

MED ARH 2011; 65(6): 373-374 Case report

molecules of the denatured protein. The


trypsin and chymotrypsin as the endopeptidases tear the other polypeptide
chains into smaller ones with their proteolytic activity on the place of the pathological process making them more adequate for absorption and one dragee
contains a mixture of proteolytic enzymes, trypsin and chymotrypsin with
the enzyme activity of 100,000 AJ. The
treatment lasted for 15 days. Three years
after the conducted therapy the patient
was in complete remission.

4. Conclusion
We believe that Chimoral Forte can
stand as an effective choice of treatment
for Cheilitis granulomatosa and can
thus help in avoiding the side-effects
of corticosteroids and/or other more
aggressive treatment procedures.
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