- Email: [email protected]
Aphthous stomatitis with involvement of attached gingiva
Aphthous stomatitis with involvement of attached gingiva Greg A. Mintz, D.M.D.,* CASE WESTERN
RESERVE
and Eric D. Smidansky,
UNIVERSITY
SCHOOL
D.M.D.,**
Cleveland, Ohio
OF DENTISTRY
Involvement of the attached gingiva by aphthous stomatitis is considered uncommon. This report presents two cases in which patients with aphthous stomatitis developed ulcers on multiple mucosal surfaces, including the attached gingiva. The gingival ulcers began on the marginal gingiva and enlarged to form Vor U-shaped ulcers on the attached gingiva. Other disorders producing gingival ulceration were excluded on the basis of history, clinical features. bacterial culture, viral culture, and biopsy with routine microscopic study as well as direct immunofluorescence. (ORAL SJRG. ORAL. MED. ORAL PATHOL. 60~122-124, 1985)
A
phthous stomatitis is characterized by the development of painful, recurrent ulcerations of the oral mucosa. Clinically, lesions appear as round or ovoid ulcers with edematous, erythematous borders. The ulcers most commonly involve labial and buccal mucosa, although other mucosal surfaces, such as the tongue, mucobuccal fold, floor of the mouth, and soft palate, may be affected.’ Involvement of keratinized mucosa bound to periosteum (attached gingiva and hard palate) is generally considered uncommon,‘-4 and aphthous stomatitis usually is not included in the differential diagnosis of gingival ulcerations. This report describes two patients who presented to Case Western Reserve University School of Dentistry with aphthous stomatitis involving multiple mucosal surfaces, including attached gingiva. CASE REPORTS CASE 1
A 34-year-old white woman sought evaluation of severely painful oral ulcers which had been recurring for the past 6 years. Examination revealed multiple ulcers, varying in size from 0.2 to 1.O cm, each with a gray necrotic base and an erythematous border; no vesicles were noted. Most of the ulcers involved labial and buccal mucosa; however, several were present on mandibular and maxillary facial gingiva as well as palatal gingiva (Fig. 1). The patient complained of joint pain which began 4 years earlier following an accident in which she sustained a fracture of the spine. Her symptoms included morning *Assistant **Clinical
122
Professor, Instructor,
Department Department
of Oral Diagnosis. of Periodontology
Fig.
1. Uicer of palatal gingiva in Patient 1
stiffness and aching of the knees, hands, and shoulders. Laboratory findings, including a complete blood count with differential and SMA 20, were within normal limits. Tests for antinuclear antibodies and rheumatoid factor were negative. Initial examination by a rheumatologist led to consideration of a seronegative arthropathy, such as Reiter’s syndrome, to account for the patient’s joint symptoms. However, follow-up examinations failed to reveal any evidence of joint patholsis or other components
Volume 60 Number 1
Fig.
Aphthous stomatitis with involvement of attached gingiva
2. Ulcer of facial gingiva in Patient 2.
of Reiter’s syndrome, and the patient was diagnosed as having fibrositis. Evaluation of the patient’s oral ulcerations included a biopsy, which revealed a nonspecific mucosal ulcer. An additional specimen submitted for direct immunofluorescencefailed to reveal the presence of complement, immunoglobulin, or fibrinogen. Bacterial culture revealed normal oral flora. Viral culture taken within 48 hours of onset of ulceration failed to detect herpes simplex virus. The patient was followed for more than a year, and during that time multiple ulcers developed at various sites on the attached gingiva. The ulcers began on marginal gingiva and enlarged to form a V- or U-shaped ulcer on attached gingiva. The patient kept a daily log, recording any episode of oral ulceration, and the frequency and severity of her gingival ulcers paralleled the frequency and severity of ulcers located elsewhere in the oral cavity. All ulcers healed in 1 to 3 weeks without scarring. The lesions were unresponsive to topical steroids, but 10 mg prednisone daily produced relief of symptoms and a decrease in the frequency of ulceration. Diagnosis: Aphthous stomatitis with involvement of attached gingiva. CASE 2
A 25-year-old white woman had a l-year history of slightly painful, recurrent ulcers of the gingiva. During this time the patient noted approximately twelve episodes of ulceration involving multiple sites on the attached gingiva. The ulcers healed completely in 1 to 3 weeks. Examination revealed multiple ulcers of maxillary and mandibular attached gingiva (Fig. 2). The ulcers were Vor U-shaped and began on marginal gingiva, sometimes involving the interdental papilla. In addition, there were several discrete ulcers with erythematous halos on the labial and buccal mucosa. When questioned about these other ulcers, the patient replied that she routinely developed “canker sores.” The medical history was unremarkable, and routine laboratory findings were within normal limits. A biopsy
123
Fig. 3. Aphthous ulcer originating on alveolar mucosa that enlarged to secondarily involve the attached gingiva.
revealed a nonspecific mucosal ulcer, and a specimen submitted for direct immunofluorescence was negative for complement, immunoglobulin, and fibrinogen. Culture for herpes simplex virus taken soon after the onset of ulceration was negative. The patient has been followed for several months and has not required steroid therapy. Diagnosis: Aphthous stomatitis with involvement of attached gingiva. DISCUSSION
While involvement of attached gingiva by aphthous stomatitis is generally considered uncommon,’ few studies have actually documented the frequency of aphthous ulcers on the gingiva. One study,5 which involved clinical examinations of 300 students with a history of aphthous stomatitis attending professional schools at the University of Pennsylvania, revealed that approximately 6% of the ulcers identified were located on the “gums.” Another study,6 at the National Institute of Dental Research, of 62 patients with aphthous stomatitis revealed that in 35% of the patients lesions developed on the gingiva. Neither report described the clinical appearance of the gingival lesions. One problem in evaluating reports of gingival involvement by aphthous stomatitis is that lesions arising on the gingiva (primary gingival involvement) have not been clearly separated from lesions that arise on alveolar mucosa and, with enlargement, extend to involve the attached gingiva (secondary gingival involvement). This latter pattern of involvement, which is illustrated in Fig. 3 and Fig. 4, a, occurs relatively frequently in the experience of one author (G.M.) of this report and presents little difficulty in diagnosis, as the lesions are otherwise typical of aphthous ulcers.
124
Mntz
and Smidansky
Oral Surg. July, 1985
Fig. 4. a, Secondary involvement of attached gingiva by ulcer originating on alveolar mucosa. b, Pattern of primary involvement of gingiva experienced by Patient 1 and Patient 2.
The two patients presented in this report had primary involvement of the gingiva by aphthous stomatitis, with multiple recurrent ulcers arising at various sites along the gingiva. Their lesions began on the marginal gingiva and enlarged to form V- or U-shaped ulcers on attached gingiva, as illustrated in Fig. 4, b. This pattern of gingival ulceration has not been previously described in aphthous stomatitis. While the patients were being evaluated, the possibility was raised that the gingival ulcers, because of their atypical appearance, might represent a separate disorder from aphthous stomatitis. However, the gingival ulcers, like the typical aphthous ulcers on other mucosal surfaces, were recurrent and involved multiple sites along the gingiva. In addition, in Patient 1 the severity and frequency of the gingival ulcers clearly paralleled the frequency and severity of ulcers on other mucosal surfaces, including the response of the lesions to systemic steroids. These findings strongly suggested that the gingival ulcers in these patients were a manifestation of aphthous stomatitis. A number of different disorders can produce ulcerations of the gingiva, and these were excluded by history (traumatic ulcerations), bacterial culture (streptococcal gingivostomatitis), and biopsy with routine microscopy and direct immunofluorescence (pemphigus vulgaris and benign mucous membrane pemphigoid, also known as a cicatricial pemphigoid). Vesicles were never noted, and cultures for herpes simplex failed to detect the virus. One disorder which bears some resemblance to the
lesions described in this report is the idiopathic gingival ulceration described by Jacobsen and associates’ who reported patients with ulceration of the attached gingiva unresponsive to antibiotics or steroids. Four of the patients in their report experienced multiple episodes of ulceration (maximum was five episodes).Although there has been a suggestion that these ulcers were traumatic in origin6 their etiology remains unknown. The patients in the present report, unlike those with idiopathic gingival ulceration, had typical aphthous ulcers at other sites on the mucosa. In addition, the gingival ulcerations described in the present report differed in duration (1 to 3 weeks) from the duration of ulceration in patients with idiopathic gingival ulceration (3 to 32 weeks). Finally, the one patient in the present report who was treated with steroids responded favorably, in contrast to the response of patients with idiopathic gingival ulceration. This report has documented two casesof aphthous stomatitis with primary involvement of attached gingiva in a previously unreported pattern of V- or U-shaped ulcers. A variety of diagnostic techniques were used to exclude other disorders producing gingival ulceration. REFERENCES Antoon JW, Miller RL: Aphthous ulcers-a review of the literature on etiology, pathogenesis, diagnosis, and treatment. J Am Dent Assoc 101: 803-808. 1980. Barile MF, Graykowski EA: Primary herpes, recurrent labial herpes, recurrent aphthae, and periadenitis aphthac: a review with some observations. J DC Dent Sot 38: 7-15, 1963. Powell FC, Rogers RS 111: A practical approach to oral lesions Primary Care 10: 495-51 I, 1983. Lynch MA, Brightman VJ, Greenberg MS: Burket’s oral medicine, ed. 8, Philadelphia, 1984, J.B. Lippincott Company, p. 184. Ship II, Morris AL, Durocher RT, Burket LW: Recurrent aphthous ulcerations and recurrent herpes labialis in a professional school student population. III. Oral examinations. ORAI SLKG OR,~L Mm ORAL PATHOL 13: 1438-1444. 1960. Graykowski EA. Barile MF, Lee WB, Stanley HR Jr: Recurrent aphthous stomatitis. Clinical, therapeutic, histopathologic, and hypersensitivity aspects. JAMA 196: 129136. 1966. Jacobsen PL, Olson JA, Silverman S Jr, Merrell P: Idiopathic gingival ulcerations: ten cases of a previously unreported entity. ORAL SURC ORAL MED ORAI. PATHOI. 50: 517-519. 1980. Schiodt M: Letter. Traumatic lesions of the gingiva provoked by toothbrushing. ORAL SURC ORAL MED ORAL PATHOL 52: 261, 1981. Reprint requests to: Dr. Greg A. Mintz Department of Oral Diagnosis Case Western Reserve Universitv School of Dentistry 2123 Abington Rd. Cleveland, OH 44106
RESERVE
and Eric D. Smidansky,
UNIVERSITY
SCHOOL
D.M.D.,**
Cleveland, Ohio
OF DENTISTRY
Involvement of the attached gingiva by aphthous stomatitis is considered uncommon. This report presents two cases in which patients with aphthous stomatitis developed ulcers on multiple mucosal surfaces, including the attached gingiva. The gingival ulcers began on the marginal gingiva and enlarged to form Vor U-shaped ulcers on the attached gingiva. Other disorders producing gingival ulceration were excluded on the basis of history, clinical features. bacterial culture, viral culture, and biopsy with routine microscopic study as well as direct immunofluorescence. (ORAL SJRG. ORAL. MED. ORAL PATHOL. 60~122-124, 1985)
A
phthous stomatitis is characterized by the development of painful, recurrent ulcerations of the oral mucosa. Clinically, lesions appear as round or ovoid ulcers with edematous, erythematous borders. The ulcers most commonly involve labial and buccal mucosa, although other mucosal surfaces, such as the tongue, mucobuccal fold, floor of the mouth, and soft palate, may be affected.’ Involvement of keratinized mucosa bound to periosteum (attached gingiva and hard palate) is generally considered uncommon,‘-4 and aphthous stomatitis usually is not included in the differential diagnosis of gingival ulcerations. This report describes two patients who presented to Case Western Reserve University School of Dentistry with aphthous stomatitis involving multiple mucosal surfaces, including attached gingiva. CASE REPORTS CASE 1
A 34-year-old white woman sought evaluation of severely painful oral ulcers which had been recurring for the past 6 years. Examination revealed multiple ulcers, varying in size from 0.2 to 1.O cm, each with a gray necrotic base and an erythematous border; no vesicles were noted. Most of the ulcers involved labial and buccal mucosa; however, several were present on mandibular and maxillary facial gingiva as well as palatal gingiva (Fig. 1). The patient complained of joint pain which began 4 years earlier following an accident in which she sustained a fracture of the spine. Her symptoms included morning *Assistant **Clinical
122
Professor, Instructor,
Department Department
of Oral Diagnosis. of Periodontology
Fig.
1. Uicer of palatal gingiva in Patient 1
stiffness and aching of the knees, hands, and shoulders. Laboratory findings, including a complete blood count with differential and SMA 20, were within normal limits. Tests for antinuclear antibodies and rheumatoid factor were negative. Initial examination by a rheumatologist led to consideration of a seronegative arthropathy, such as Reiter’s syndrome, to account for the patient’s joint symptoms. However, follow-up examinations failed to reveal any evidence of joint patholsis or other components
Volume 60 Number 1
Fig.
Aphthous stomatitis with involvement of attached gingiva
2. Ulcer of facial gingiva in Patient 2.
of Reiter’s syndrome, and the patient was diagnosed as having fibrositis. Evaluation of the patient’s oral ulcerations included a biopsy, which revealed a nonspecific mucosal ulcer. An additional specimen submitted for direct immunofluorescencefailed to reveal the presence of complement, immunoglobulin, or fibrinogen. Bacterial culture revealed normal oral flora. Viral culture taken within 48 hours of onset of ulceration failed to detect herpes simplex virus. The patient was followed for more than a year, and during that time multiple ulcers developed at various sites on the attached gingiva. The ulcers began on marginal gingiva and enlarged to form a V- or U-shaped ulcer on attached gingiva. The patient kept a daily log, recording any episode of oral ulceration, and the frequency and severity of her gingival ulcers paralleled the frequency and severity of ulcers located elsewhere in the oral cavity. All ulcers healed in 1 to 3 weeks without scarring. The lesions were unresponsive to topical steroids, but 10 mg prednisone daily produced relief of symptoms and a decrease in the frequency of ulceration. Diagnosis: Aphthous stomatitis with involvement of attached gingiva. CASE 2
A 25-year-old white woman had a l-year history of slightly painful, recurrent ulcers of the gingiva. During this time the patient noted approximately twelve episodes of ulceration involving multiple sites on the attached gingiva. The ulcers healed completely in 1 to 3 weeks. Examination revealed multiple ulcers of maxillary and mandibular attached gingiva (Fig. 2). The ulcers were Vor U-shaped and began on marginal gingiva, sometimes involving the interdental papilla. In addition, there were several discrete ulcers with erythematous halos on the labial and buccal mucosa. When questioned about these other ulcers, the patient replied that she routinely developed “canker sores.” The medical history was unremarkable, and routine laboratory findings were within normal limits. A biopsy
123
Fig. 3. Aphthous ulcer originating on alveolar mucosa that enlarged to secondarily involve the attached gingiva.
revealed a nonspecific mucosal ulcer, and a specimen submitted for direct immunofluorescence was negative for complement, immunoglobulin, and fibrinogen. Culture for herpes simplex virus taken soon after the onset of ulceration was negative. The patient has been followed for several months and has not required steroid therapy. Diagnosis: Aphthous stomatitis with involvement of attached gingiva. DISCUSSION
While involvement of attached gingiva by aphthous stomatitis is generally considered uncommon,’ few studies have actually documented the frequency of aphthous ulcers on the gingiva. One study,5 which involved clinical examinations of 300 students with a history of aphthous stomatitis attending professional schools at the University of Pennsylvania, revealed that approximately 6% of the ulcers identified were located on the “gums.” Another study,6 at the National Institute of Dental Research, of 62 patients with aphthous stomatitis revealed that in 35% of the patients lesions developed on the gingiva. Neither report described the clinical appearance of the gingival lesions. One problem in evaluating reports of gingival involvement by aphthous stomatitis is that lesions arising on the gingiva (primary gingival involvement) have not been clearly separated from lesions that arise on alveolar mucosa and, with enlargement, extend to involve the attached gingiva (secondary gingival involvement). This latter pattern of involvement, which is illustrated in Fig. 3 and Fig. 4, a, occurs relatively frequently in the experience of one author (G.M.) of this report and presents little difficulty in diagnosis, as the lesions are otherwise typical of aphthous ulcers.
124
Mntz
and Smidansky
Oral Surg. July, 1985
Fig. 4. a, Secondary involvement of attached gingiva by ulcer originating on alveolar mucosa. b, Pattern of primary involvement of gingiva experienced by Patient 1 and Patient 2.
The two patients presented in this report had primary involvement of the gingiva by aphthous stomatitis, with multiple recurrent ulcers arising at various sites along the gingiva. Their lesions began on the marginal gingiva and enlarged to form V- or U-shaped ulcers on attached gingiva, as illustrated in Fig. 4, b. This pattern of gingival ulceration has not been previously described in aphthous stomatitis. While the patients were being evaluated, the possibility was raised that the gingival ulcers, because of their atypical appearance, might represent a separate disorder from aphthous stomatitis. However, the gingival ulcers, like the typical aphthous ulcers on other mucosal surfaces, were recurrent and involved multiple sites along the gingiva. In addition, in Patient 1 the severity and frequency of the gingival ulcers clearly paralleled the frequency and severity of ulcers on other mucosal surfaces, including the response of the lesions to systemic steroids. These findings strongly suggested that the gingival ulcers in these patients were a manifestation of aphthous stomatitis. A number of different disorders can produce ulcerations of the gingiva, and these were excluded by history (traumatic ulcerations), bacterial culture (streptococcal gingivostomatitis), and biopsy with routine microscopy and direct immunofluorescence (pemphigus vulgaris and benign mucous membrane pemphigoid, also known as a cicatricial pemphigoid). Vesicles were never noted, and cultures for herpes simplex failed to detect the virus. One disorder which bears some resemblance to the
lesions described in this report is the idiopathic gingival ulceration described by Jacobsen and associates’ who reported patients with ulceration of the attached gingiva unresponsive to antibiotics or steroids. Four of the patients in their report experienced multiple episodes of ulceration (maximum was five episodes).Although there has been a suggestion that these ulcers were traumatic in origin6 their etiology remains unknown. The patients in the present report, unlike those with idiopathic gingival ulceration, had typical aphthous ulcers at other sites on the mucosa. In addition, the gingival ulcerations described in the present report differed in duration (1 to 3 weeks) from the duration of ulceration in patients with idiopathic gingival ulceration (3 to 32 weeks). Finally, the one patient in the present report who was treated with steroids responded favorably, in contrast to the response of patients with idiopathic gingival ulceration. This report has documented two casesof aphthous stomatitis with primary involvement of attached gingiva in a previously unreported pattern of V- or U-shaped ulcers. A variety of diagnostic techniques were used to exclude other disorders producing gingival ulceration. REFERENCES Antoon JW, Miller RL: Aphthous ulcers-a review of the literature on etiology, pathogenesis, diagnosis, and treatment. J Am Dent Assoc 101: 803-808. 1980. Barile MF, Graykowski EA: Primary herpes, recurrent labial herpes, recurrent aphthae, and periadenitis aphthac: a review with some observations. J DC Dent Sot 38: 7-15, 1963. Powell FC, Rogers RS 111: A practical approach to oral lesions Primary Care 10: 495-51 I, 1983. Lynch MA, Brightman VJ, Greenberg MS: Burket’s oral medicine, ed. 8, Philadelphia, 1984, J.B. Lippincott Company, p. 184. Ship II, Morris AL, Durocher RT, Burket LW: Recurrent aphthous ulcerations and recurrent herpes labialis in a professional school student population. III. Oral examinations. ORAI SLKG OR,~L Mm ORAL PATHOL 13: 1438-1444. 1960. Graykowski EA. Barile MF, Lee WB, Stanley HR Jr: Recurrent aphthous stomatitis. Clinical, therapeutic, histopathologic, and hypersensitivity aspects. JAMA 196: 129136. 1966. Jacobsen PL, Olson JA, Silverman S Jr, Merrell P: Idiopathic gingival ulcerations: ten cases of a previously unreported entity. ORAL SURC ORAL MED ORAI. PATHOI. 50: 517-519. 1980. Schiodt M: Letter. Traumatic lesions of the gingiva provoked by toothbrushing. ORAL SURC ORAL MED ORAL PATHOL 52: 261, 1981. Reprint requests to: Dr. Greg A. Mintz Department of Oral Diagnosis Case Western Reserve Universitv School of Dentistry 2123 Abington Rd. Cleveland, OH 44106