Necrotizing Sialometaplasia Report of Eugene
N.
a
Case
Myers, MD; Murat Bankaci, MD; E. L. Barnes, Jr, MD
sialometaplasia occurred 46-year-old woman. This is an uncommon benign disease that typically begins with an ulcerated lesion on the hard palate and surrounding tissues. Clinical and microscopic findings show obvious similarity with a malignant neoplasm. Necrotizing sialometaplasia is a benign minor salivary gland disease. It is important since it may easily be confused with \s=b\ Necrotizing
in
a
squamous cell carcinoma and mucoepidermoid carcinoma.
(Arch Otolaryngol 101:628-629, 1975)
past During "new" benign disease the
two
years,
a
of minor salivary glands has been described.1 Necrotizing sialometaplasia is an un¬ common disease that typically begins with an ulcerated lesion on the hard palate and surrounding tissues. The cause of this disease, which is seen only in adults, is unknown. In all re¬ ported cases, an ulcerated lesion was present on the palate and surround¬ ing tissues.1-2 Clinically, it has no spe¬ cific diagnostic features. Microscopi¬ cally, lobular necrosis and extensive
metaplasia of minor sali¬ vary glands are consistent findings.1 It is of utmost importance to recog¬ nize this benign disease since its clini¬ cal and microscopie findings simulate a malignant neoplasm, especially mucoepidermoid carcinoma or squamous squamous
cell carcinoma.
REPORT OF A CASE In August 1974, a 46-year-old woman was referred to the Otolaryngology Service with a lesion on her palate of four weeks duration that was associated with pain in her left ear. There were no other otologie complaints. There was no history of smok¬ ing or ethyl alcohol use. Physical examina-
tion revealed a 1 2-cm ulcerated lesion at the junction of the hard and soft palate on the left side, adjacent to the midline, with
erythematous margins anteriorly (Fig 1). No cervical lymphadenopathic features were noted. The patient underwent an excisional biopsy of the palatal lesion and the histopathologic diagnosis was mucoepider-
moid carcinoma. She was then referred to the Otolaryngology Service for definitive surgery. A three-dimensional excision with an en bloc removal of the posterior part of the hard palate, anterior part of the soft palate, and left alveolar ridge was done with the patient under general anesthesia. Local mucosal flaps were used to resurface the edges of the palatal defect. Surgical margins were reported to be free of tumor.
Fig 1.—Intraoral view showing ulcerative lesion gins.
Accepted for publication May 16, 1975. From the Department of Otolaryngology, Eye and Ear Hospital (Drs Myers and Bankaci), and the departments of otolaryngology (Drs Myers and Bankaci) and pathology (Dr Barnes), University of Pittsburgh School of Medicine, Pitts-
burgh. Reprint requests to the Department of Otolaryngology, Eye and Ear Hospital, Pittsburgh, PA 15213 (Dr Myers).
Downloaded From: http://archotol.jamanetwork.com/ by a Michigan State University User on 06/15/2015
of hard
palate and erythematous
mar¬
Fig 2.—Ulcerated mucosa and ischemie acini with many exhi¬ biting disruptions of their walls and stornai release of mucus and secondary inflammation (hematoxylin-eosin, original magnifica¬
tion
A
Fig 3.—Adjacent minor salivary gland lobules exhibiting extensive squamous metaplasia of acini and ducts (hematoxylin-eosin, original magnification 4).
4).
temporary obturator prosthesis
was
in¬
serted.
HISTOPATHOLOGIC FINDINGS
The definitive surgical specimen consisted of a 5.0x4.0xl.7-cm seg¬ ment of palate with attached bone. A lx2-cm ulcer occupied the center of the tissue and extended to a depth of 0.7 cm. The base of the ulcer was "clean," and the margins were er¬ ythematous and only mildly indu¬ rated. Light microscopic examination of hematoxylin-eosin-stained sections revealed the mucosal margins to be acanthotic with areas of pseudoepithelomatous hyperplasia. The base was formed of necrotic debris, fibrin, and neutrophils overlying granula¬ tion tissue. Subadjacent to the ulcer, lobules of ischemie minor salivary glands exhibited focal dissolution of acinar walls with stromal release of mucin. The free mucin stimulated a brisk inflammatory reaction that con¬ sisted of neutrophils and foamy his¬ tiocytes with a granulation tissue re¬ sponse (Fig 2). The most prominent change, however, was extensive squa¬ mous metaplasia of salivary acini and ducts occasionally seen in the is¬ chemie lobules, but more commonly present in bordering nonischemic but inflamed salivary lobules (Fig 3). The metaplastic squamous cells were, for the most part, uniform and bland, al¬ though occasional "reactive" nuclear changes and infrequent normal mi¬ toses were observed. Despite the
extensive ulcération and
in¬ ob¬ flammatory reaction, low-power servation revealed that the basic lobular salivary gland architecture was intact. Some of the arteries did reveal a slight medial hypertrophy, but there were no intimai changes. A review of the original biopsy speci¬ men revealed many of the micro¬ scopic features as noted previously. severe
COMMENT
reported case of necrotizing sialometaplasia. Of the 12 previously reported cases all but one initiated with an ulcer of the hard pal¬ ate and surrounding tissues.1-2 The dis¬ This is the 13th
ease
occurred in adults of ages vary¬
ing from 23 to 66 years old. Eight of the 12 patients were over 40 years of age; nine patients were men, and
three were women. Nine of the 12 pa¬ tients were white and one was black. The race of two patients was un¬ known. This disease is reported to be slow-healing. Two patients had spon¬ taneous healing and one patient had complete excision. Six patients had incomplete excision with surgical margins showing presence of disease. All patients were healed and free of disease on follow-up varying from four months to eight years after
Abrams et al1 were the first to de¬ scribe the microscopic appearance of this lesion as follows: (1) mucosal ulcération of palate with pseudoepitheliomatous hyperplasia; (2) ischemie lobular necrosis of salivary glands; (3) dissolution of acinar walls with mu¬ cous release and secondary inflamma¬ tory and granulation tissue response; (4) extensive squamous metaplasia of salivary acini and ducts found almost invariably in multiple lobules; and (5) relatively intact salivary lobular ar¬ chitecture despite the extensive ne¬ crosis and inflammation.
Necrotizing sialometaplasia should
be considered in the differential diag¬ nosis of ulcerative lesions on the pal¬ ate, since it is frequently confused with malignant tumors of minor sali¬ vary glands, especially mucoepidermoid carcinoma and squamous cell carcinoma. A definite diagnosis may be estab¬ lished only with microscopic examina¬ tion of the lesion. Confirming the proper diagnosis eliminates the need for unnecessary surgery since all of the reported patients healed com¬ pletely regardless of the mode of therapy—complete excision, incom¬ plete excision, or no surgical treat¬ ment.
treatment.
Necrotizing sialometaplasia is a disease of unknown cause. It typically begins with an ulcerated lesion on the palate and surrounding tissues. It oc¬ curs exclusively in adults, especially in those
over
40 years of age.
References 1. Abrams AM, Melrose RJ, Howell FV: Necrotizing sialometaplasia: A disease simulating malignancy. Cancer 31:130-135, 1973. 2. Dunlap CL, Barker BF: Necrotizing sialometaplasia: Report of five additional cases. Oral Surg 37:722-727, 1974
Downloaded From: http://archotol.jamanetwork.com/ by a Michigan State University User on 06/15/2015
N.
a
Case
Myers, MD; Murat Bankaci, MD; E. L. Barnes, Jr, MD
sialometaplasia occurred 46-year-old woman. This is an uncommon benign disease that typically begins with an ulcerated lesion on the hard palate and surrounding tissues. Clinical and microscopic findings show obvious similarity with a malignant neoplasm. Necrotizing sialometaplasia is a benign minor salivary gland disease. It is important since it may easily be confused with \s=b\ Necrotizing
in
a
squamous cell carcinoma and mucoepidermoid carcinoma.
(Arch Otolaryngol 101:628-629, 1975)
past During "new" benign disease the
two
years,
a
of minor salivary glands has been described.1 Necrotizing sialometaplasia is an un¬ common disease that typically begins with an ulcerated lesion on the hard palate and surrounding tissues. The cause of this disease, which is seen only in adults, is unknown. In all re¬ ported cases, an ulcerated lesion was present on the palate and surround¬ ing tissues.1-2 Clinically, it has no spe¬ cific diagnostic features. Microscopi¬ cally, lobular necrosis and extensive
metaplasia of minor sali¬ vary glands are consistent findings.1 It is of utmost importance to recog¬ nize this benign disease since its clini¬ cal and microscopie findings simulate a malignant neoplasm, especially mucoepidermoid carcinoma or squamous squamous
cell carcinoma.
REPORT OF A CASE In August 1974, a 46-year-old woman was referred to the Otolaryngology Service with a lesion on her palate of four weeks duration that was associated with pain in her left ear. There were no other otologie complaints. There was no history of smok¬ ing or ethyl alcohol use. Physical examina-
tion revealed a 1 2-cm ulcerated lesion at the junction of the hard and soft palate on the left side, adjacent to the midline, with
erythematous margins anteriorly (Fig 1). No cervical lymphadenopathic features were noted. The patient underwent an excisional biopsy of the palatal lesion and the histopathologic diagnosis was mucoepider-
moid carcinoma. She was then referred to the Otolaryngology Service for definitive surgery. A three-dimensional excision with an en bloc removal of the posterior part of the hard palate, anterior part of the soft palate, and left alveolar ridge was done with the patient under general anesthesia. Local mucosal flaps were used to resurface the edges of the palatal defect. Surgical margins were reported to be free of tumor.
Fig 1.—Intraoral view showing ulcerative lesion gins.
Accepted for publication May 16, 1975. From the Department of Otolaryngology, Eye and Ear Hospital (Drs Myers and Bankaci), and the departments of otolaryngology (Drs Myers and Bankaci) and pathology (Dr Barnes), University of Pittsburgh School of Medicine, Pitts-
burgh. Reprint requests to the Department of Otolaryngology, Eye and Ear Hospital, Pittsburgh, PA 15213 (Dr Myers).
Downloaded From: http://archotol.jamanetwork.com/ by a Michigan State University User on 06/15/2015
of hard
palate and erythematous
mar¬
Fig 2.—Ulcerated mucosa and ischemie acini with many exhi¬ biting disruptions of their walls and stornai release of mucus and secondary inflammation (hematoxylin-eosin, original magnifica¬
tion
A
Fig 3.—Adjacent minor salivary gland lobules exhibiting extensive squamous metaplasia of acini and ducts (hematoxylin-eosin, original magnification 4).
4).
temporary obturator prosthesis
was
in¬
serted.
HISTOPATHOLOGIC FINDINGS
The definitive surgical specimen consisted of a 5.0x4.0xl.7-cm seg¬ ment of palate with attached bone. A lx2-cm ulcer occupied the center of the tissue and extended to a depth of 0.7 cm. The base of the ulcer was "clean," and the margins were er¬ ythematous and only mildly indu¬ rated. Light microscopic examination of hematoxylin-eosin-stained sections revealed the mucosal margins to be acanthotic with areas of pseudoepithelomatous hyperplasia. The base was formed of necrotic debris, fibrin, and neutrophils overlying granula¬ tion tissue. Subadjacent to the ulcer, lobules of ischemie minor salivary glands exhibited focal dissolution of acinar walls with stromal release of mucin. The free mucin stimulated a brisk inflammatory reaction that con¬ sisted of neutrophils and foamy his¬ tiocytes with a granulation tissue re¬ sponse (Fig 2). The most prominent change, however, was extensive squa¬ mous metaplasia of salivary acini and ducts occasionally seen in the is¬ chemie lobules, but more commonly present in bordering nonischemic but inflamed salivary lobules (Fig 3). The metaplastic squamous cells were, for the most part, uniform and bland, al¬ though occasional "reactive" nuclear changes and infrequent normal mi¬ toses were observed. Despite the
extensive ulcération and
in¬ ob¬ flammatory reaction, low-power servation revealed that the basic lobular salivary gland architecture was intact. Some of the arteries did reveal a slight medial hypertrophy, but there were no intimai changes. A review of the original biopsy speci¬ men revealed many of the micro¬ scopic features as noted previously. severe
COMMENT
reported case of necrotizing sialometaplasia. Of the 12 previously reported cases all but one initiated with an ulcer of the hard pal¬ ate and surrounding tissues.1-2 The dis¬ This is the 13th
ease
occurred in adults of ages vary¬
ing from 23 to 66 years old. Eight of the 12 patients were over 40 years of age; nine patients were men, and
three were women. Nine of the 12 pa¬ tients were white and one was black. The race of two patients was un¬ known. This disease is reported to be slow-healing. Two patients had spon¬ taneous healing and one patient had complete excision. Six patients had incomplete excision with surgical margins showing presence of disease. All patients were healed and free of disease on follow-up varying from four months to eight years after
Abrams et al1 were the first to de¬ scribe the microscopic appearance of this lesion as follows: (1) mucosal ulcération of palate with pseudoepitheliomatous hyperplasia; (2) ischemie lobular necrosis of salivary glands; (3) dissolution of acinar walls with mu¬ cous release and secondary inflamma¬ tory and granulation tissue response; (4) extensive squamous metaplasia of salivary acini and ducts found almost invariably in multiple lobules; and (5) relatively intact salivary lobular ar¬ chitecture despite the extensive ne¬ crosis and inflammation.
Necrotizing sialometaplasia should
be considered in the differential diag¬ nosis of ulcerative lesions on the pal¬ ate, since it is frequently confused with malignant tumors of minor sali¬ vary glands, especially mucoepidermoid carcinoma and squamous cell carcinoma. A definite diagnosis may be estab¬ lished only with microscopic examina¬ tion of the lesion. Confirming the proper diagnosis eliminates the need for unnecessary surgery since all of the reported patients healed com¬ pletely regardless of the mode of therapy—complete excision, incom¬ plete excision, or no surgical treat¬ ment.
treatment.
Necrotizing sialometaplasia is a disease of unknown cause. It typically begins with an ulcerated lesion on the palate and surrounding tissues. It oc¬ curs exclusively in adults, especially in those
over
40 years of age.
References 1. Abrams AM, Melrose RJ, Howell FV: Necrotizing sialometaplasia: A disease simulating malignancy. Cancer 31:130-135, 1973. 2. Dunlap CL, Barker BF: Necrotizing sialometaplasia: Report of five additional cases. Oral Surg 37:722-727, 1974
Downloaded From: http://archotol.jamanetwork.com/ by a Michigan State University User on 06/15/2015
