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Angular cheilosis and macroglossia
Associate Editor ‘I~I?Z3TER 92. CAHN
epsti of a Case ~RNTwI’
~AIxw,~
CAPTAIN,
USAF
(DC),
AND
HAROLD
R.
PIERCE,“”
‘CJSAF (MC)
CAPTAIN,
Introduction ERLMXIE or angular cheilosis is an inflammatory and atrophic condition characterized by cracking and fissuring at the corner of the mouth and erosion of the mucous membranes. This clinical entity may be caused by a number of factors, including nutritional deficiencies such as avitaminoses, and specifically by absence or nona,bsorption of vitamin B,. Chronic inflammation, maceration, secondary infection, and degenerative changes usually induced by a collapse of the occlusion and decrease of the normal vertical intermaxillary space are other predisposing factors. Riboflavin deficiency is best known as a cause of angular cheilosis and can be produced experimentally by a pellagra diet.l This particular deficiency generally presents eye disturbances such as lacrimation, conjunctivitis, and burning of the eyes, as well as atrophic tongue changes. This type of vitamin deficiency generaBy responds rapidly to riboflavin therapy. In those cases where the cracking of the commissures slants vertically and does not radiate from the labial commissures in a fanlike fashion, a decrease of the vertical dimension should be suspected. The following case presented a diagnostic problem and points out the relationship between macroglossia and angular cheilosis. ~I____From the 1600th USAF “Chief of Oral Surgery. “*Chief of Derma.tology.
Hospital,
Westover
PO0
Air
Force
Base,
Massachusetts.
AN0 ULAB
CHIBLOSIS
.101
ANL) MACIZOGLOSSIA
Case Report A 21.year-old white man was first examined in the dental clinic at Westover Air Force .Kase Ilospital on Aug. 3, 1953. Be complained of cracking, redness, and soreness about the corners of his mouth. The onset of the patient’s symptoms occurred about March, 1953. There was no itching, burning, or severe pain, but an annoying soreness began during the routine process of conversation and eating, at which times the labial commissures became fissured. The patient appeared to be in good health, and the past The dietary intake was normal, and a medical n.nd dental history was noncontributory. one-week dietary intake analysis did not reveal any mincrxl, protein, or vitamin deficiencies indicative of gross malnutrition. Examination.-The patient was a well-developed, well-nourished white man in no The lesions acute distress, presenting bilateral angular choilosis at the labial commissures. were fissured, radiating in a fanlike fashion from the corner of the mouth, and appearing to be shallow, well delimited, and covered by a crusty, yellow discharge. Some erythema was evident at the periphery of the lesions, and the inflammatory reaction appeared to be greatest toward the mucosal aspect of the lips with a slight extension onto the skin near the vermilion border. The direction of the Rssures was along the lip following the natural folds of the skin.
Fig. l.-Angular
cheilosis
in patient
seen initially
on hug.
3, 1953
There was no lymphadenopathy. The oral mucous membranes were otherwise normal. The patient was partially edentulous, and the examination of the remaining dentition and occlusion revealed a decrease of the normal vertical dimension, The general physicai examination revealed no abnormality. Laboratory tests were all within normal limits, and the bacteriologic smears of the lesion were negative for specific organisms. All findings pointed to a traumatic lesion, produced by maceration of the labial commissures by the excessive folds resulting from a decrease in the vertical dimension. Although this was considered. the determining factor in the ctiological diagnosis of this cheilosis, the possibility of a vitamin B complex deficiency or, more specifically, a riboflavin deficiency was considered. Treatment.--As the patient needed a new upper full also aimed at restoring the vertical dimension.
denture,
the prosthetic
traatment
On Sept. 13, 3953, the upper denture was inserted. Following a detailed dietarg his tory, a well-balanced diet was prescribed. with natural vitamin supplements. No specific substitution therapy was recommended at this time. After insertion of the dentures, which restored a normal vertical dimension, the condit,ion of the lips improved for a few months, but did not subside completely. The patient noted that, although the lesions disappeared for about eight weeks, zome redness of the labial commissures remained conWhen the patient was seen again on Oct. 22, 1953, the angular cheilosis was stantly.
was
Fig.
3.
Fig.
4.
Rig. 3.-Recurrence of the lesions on Dec. 4, 1953. Fig. 4.-Acute recurrence of angular lesions, about Evidence of congenital macroglossia. discontinued.
one
month
after
systemic
therapy
AXGULAR
CHEILOSIS
AND
103
WIACROGLOSSlA
again present with its original signs and symptoms. It was felt that a riboflavin deficiency per&he would be a tenable diagnosis at this time, and riboflavin (10 mg. four times a day for three weeks) was prescribed orally. On Nov. 6, 1953, the lesions seemed to have improved somewhat, and two weeks later they had completely disappeared. A definitive cure was not achieved, since the patient returned on December 4 with another episode of angular cheilosis. After consultation with one of us and a complete physical examination, a trial treatment of injectable B complex and liver extract was instituted. The patient received 1 C.C. of crude liver extract three times a week for two weeks and then once weekly for two months. It was felt that the patient’s progress was significaut during this period. A well-balanced diet was maintained, supplemented by multivitamin capsules. On March 28, 1954, the patient returned to the dental clinic with similar complaints. A that the patient frequently more careful history was taken at this time which revealed When this fact moistened his lips during the day and presented a definite macroglossia. was called to his attention, the patient noticed that he unconsciously rested his tongue on Further, a history of mouth breathing the labial commissures when speak;-ng or eating. was found. It may be surmised from this finding that at night the tongue also rested against the corner of the lips, thus causing a constant source of irritation,
Discussion The previously men.tioned findings are in agreement with the pathogenic explanation of perl&he as origina,lly offered by the French school, wherein children afflicted with angular cheilosis licked constantly the corners of their mouth. It appears that the only explanation for this case of chronic angular cheilosis resista,rrt to all accepted methods of treatment is the etiologies1 role of Palliative treatment was recommended for the the congenital macroglossia. patient, consisting of cocoa butter applied to (the eorncrs of the lips a.nd control of the habit by re-education. Conclusion A case of chronic angular cheitosis is l)rcsented, resistant to prostheti’c and vitamin therapy. The etiological agent is a congenital macroglossia. The problems encountered in the final diagnosis and treatment of this case emphasize the importance of recording a good and thorough history, as well as conducting a searching examination, and exercising keen clinical judgment. Addendum The .following
note
.from
one of the
a.uthors,,
Captain
Kaclon,
was
received
by me.
“‘Referring to the case report .I submitted to you, I retook a biopsy specimen Sept. 14, 1954 because the patient again had a severe recurrence of angular The result of the histopathologie excheilitis and cheilosis on the left, side, aminat,ion is as follows: “Speeirneu of skin and mucosa from left labial cornmissure. There is a There is paraportion. of skin with acanthotic thickening of the epidermis. by macrophages, koratosis and elongation of rete pegs. The eorium is inliltrated lymphocytes, neutrophils, and cosinophils. An epidermis-line fissure is present i ( DingTbosis : Chronic cheilitis, cause undeterin the center of the specimen. mined. ’ ’ (L. B. C.) References
Oral Medicine; Lester W.: .J. B. Lippincott Company. 2. Thoma > Kurt %I. . Oral Pathology, 1. Burket,
Diagnosis
and Treatment,
cd. 3, St. Louis,
1950, l’he
ed. 2, Philadelphia, C. V. Mosby
1952,
Company.
epsti of a Case ~RNTwI’
~AIxw,~
CAPTAIN,
USAF
(DC),
AND
HAROLD
R.
PIERCE,“”
‘CJSAF (MC)
CAPTAIN,
Introduction ERLMXIE or angular cheilosis is an inflammatory and atrophic condition characterized by cracking and fissuring at the corner of the mouth and erosion of the mucous membranes. This clinical entity may be caused by a number of factors, including nutritional deficiencies such as avitaminoses, and specifically by absence or nona,bsorption of vitamin B,. Chronic inflammation, maceration, secondary infection, and degenerative changes usually induced by a collapse of the occlusion and decrease of the normal vertical intermaxillary space are other predisposing factors. Riboflavin deficiency is best known as a cause of angular cheilosis and can be produced experimentally by a pellagra diet.l This particular deficiency generally presents eye disturbances such as lacrimation, conjunctivitis, and burning of the eyes, as well as atrophic tongue changes. This type of vitamin deficiency generaBy responds rapidly to riboflavin therapy. In those cases where the cracking of the commissures slants vertically and does not radiate from the labial commissures in a fanlike fashion, a decrease of the vertical dimension should be suspected. The following case presented a diagnostic problem and points out the relationship between macroglossia and angular cheilosis. ~I____From the 1600th USAF “Chief of Oral Surgery. “*Chief of Derma.tology.
Hospital,
Westover
PO0
Air
Force
Base,
Massachusetts.
AN0 ULAB
CHIBLOSIS
.101
ANL) MACIZOGLOSSIA
Case Report A 21.year-old white man was first examined in the dental clinic at Westover Air Force .Kase Ilospital on Aug. 3, 1953. Be complained of cracking, redness, and soreness about the corners of his mouth. The onset of the patient’s symptoms occurred about March, 1953. There was no itching, burning, or severe pain, but an annoying soreness began during the routine process of conversation and eating, at which times the labial commissures became fissured. The patient appeared to be in good health, and the past The dietary intake was normal, and a medical n.nd dental history was noncontributory. one-week dietary intake analysis did not reveal any mincrxl, protein, or vitamin deficiencies indicative of gross malnutrition. Examination.-The patient was a well-developed, well-nourished white man in no The lesions acute distress, presenting bilateral angular choilosis at the labial commissures. were fissured, radiating in a fanlike fashion from the corner of the mouth, and appearing to be shallow, well delimited, and covered by a crusty, yellow discharge. Some erythema was evident at the periphery of the lesions, and the inflammatory reaction appeared to be greatest toward the mucosal aspect of the lips with a slight extension onto the skin near the vermilion border. The direction of the Rssures was along the lip following the natural folds of the skin.
Fig. l.-Angular
cheilosis
in patient
seen initially
on hug.
3, 1953
There was no lymphadenopathy. The oral mucous membranes were otherwise normal. The patient was partially edentulous, and the examination of the remaining dentition and occlusion revealed a decrease of the normal vertical dimension, The general physicai examination revealed no abnormality. Laboratory tests were all within normal limits, and the bacteriologic smears of the lesion were negative for specific organisms. All findings pointed to a traumatic lesion, produced by maceration of the labial commissures by the excessive folds resulting from a decrease in the vertical dimension. Although this was considered. the determining factor in the ctiological diagnosis of this cheilosis, the possibility of a vitamin B complex deficiency or, more specifically, a riboflavin deficiency was considered. Treatment.--As the patient needed a new upper full also aimed at restoring the vertical dimension.
denture,
the prosthetic
traatment
On Sept. 13, 3953, the upper denture was inserted. Following a detailed dietarg his tory, a well-balanced diet was prescribed. with natural vitamin supplements. No specific substitution therapy was recommended at this time. After insertion of the dentures, which restored a normal vertical dimension, the condit,ion of the lips improved for a few months, but did not subside completely. The patient noted that, although the lesions disappeared for about eight weeks, zome redness of the labial commissures remained conWhen the patient was seen again on Oct. 22, 1953, the angular cheilosis was stantly.
was
Fig.
3.
Fig.
4.
Rig. 3.-Recurrence of the lesions on Dec. 4, 1953. Fig. 4.-Acute recurrence of angular lesions, about Evidence of congenital macroglossia. discontinued.
one
month
after
systemic
therapy
AXGULAR
CHEILOSIS
AND
103
WIACROGLOSSlA
again present with its original signs and symptoms. It was felt that a riboflavin deficiency per&he would be a tenable diagnosis at this time, and riboflavin (10 mg. four times a day for three weeks) was prescribed orally. On Nov. 6, 1953, the lesions seemed to have improved somewhat, and two weeks later they had completely disappeared. A definitive cure was not achieved, since the patient returned on December 4 with another episode of angular cheilosis. After consultation with one of us and a complete physical examination, a trial treatment of injectable B complex and liver extract was instituted. The patient received 1 C.C. of crude liver extract three times a week for two weeks and then once weekly for two months. It was felt that the patient’s progress was significaut during this period. A well-balanced diet was maintained, supplemented by multivitamin capsules. On March 28, 1954, the patient returned to the dental clinic with similar complaints. A that the patient frequently more careful history was taken at this time which revealed When this fact moistened his lips during the day and presented a definite macroglossia. was called to his attention, the patient noticed that he unconsciously rested his tongue on Further, a history of mouth breathing the labial commissures when speak;-ng or eating. was found. It may be surmised from this finding that at night the tongue also rested against the corner of the lips, thus causing a constant source of irritation,
Discussion The previously men.tioned findings are in agreement with the pathogenic explanation of perl&he as origina,lly offered by the French school, wherein children afflicted with angular cheilosis licked constantly the corners of their mouth. It appears that the only explanation for this case of chronic angular cheilosis resista,rrt to all accepted methods of treatment is the etiologies1 role of Palliative treatment was recommended for the the congenital macroglossia. patient, consisting of cocoa butter applied to (the eorncrs of the lips a.nd control of the habit by re-education. Conclusion A case of chronic angular cheitosis is l)rcsented, resistant to prostheti’c and vitamin therapy. The etiological agent is a congenital macroglossia. The problems encountered in the final diagnosis and treatment of this case emphasize the importance of recording a good and thorough history, as well as conducting a searching examination, and exercising keen clinical judgment. Addendum The .following
note
.from
one of the
a.uthors,,
Captain
Kaclon,
was
received
by me.
“‘Referring to the case report .I submitted to you, I retook a biopsy specimen Sept. 14, 1954 because the patient again had a severe recurrence of angular The result of the histopathologie excheilitis and cheilosis on the left, side, aminat,ion is as follows: “Speeirneu of skin and mucosa from left labial cornmissure. There is a There is paraportion. of skin with acanthotic thickening of the epidermis. by macrophages, koratosis and elongation of rete pegs. The eorium is inliltrated lymphocytes, neutrophils, and cosinophils. An epidermis-line fissure is present i ( DingTbosis : Chronic cheilitis, cause undeterin the center of the specimen. mined. ’ ’ (L. B. C.) References
Oral Medicine; Lester W.: .J. B. Lippincott Company. 2. Thoma > Kurt %I. . Oral Pathology, 1. Burket,
Diagnosis
and Treatment,
cd. 3, St. Louis,
1950, l’he
ed. 2, Philadelphia, C. V. Mosby
1952,
Company.