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Benign or malignant? Case report
European Annals of Otorhinolaryngology, Head and Neck diseases (2012) 129, 275—277
Available online at
www.sciencedirect.com
CASE REPORT
Benign or malignant? Case report T. Bernardo a,∗, D. Tente b, P. Oliveira a, D. Helena a, D. Ribeiro a, S. Gerós a, A. Silva a a
ORL Department, Centro Hospitalar de Vila Nova de Gaia/Espinho-EPE, Rua Conceic¸ão Fernandes, 4434-502 Vila Nova de Gaia, Portugal b Pathology Department, Centro Hospitalar de Vila Nova de Gaia/Espinho-EPE, Rua Conceic¸ão Fernandes, 4434-502 Vila Nova de Gaia, Portugal
KEYWORDS Necrotizing sialometoplasia; Salivary glands; Oral tumors
Summary Introduction: Necrotizing sialometoplasia (NS) is a disease in which the clinical and sometimes even histological features can be confused with those of a malignant tumor, but which is in fact a self-limited inflammatory lesion of the salivary glands. Case report: The authors present a case study of a 43-year-old female patient who came to the ENT Emergency Department with a painful ulcerated lesion of the hard palate. The lesion was 8 days old and had appeared after an abdominoplasty. Given the clinical need to rule out a malignant neoplasm, a biopsy was performed: the histological result was NS. Discussion/conclusion: The authors make a literature review and stress the importance of differentiating between NS and salivary gland neoplasia. Histology is essential in case of ulcerated neoformation of the oral cavity, to adapt treatment, avoiding unsuitable approaches. © 2012 Elsevier Masson SAS. All rights reserved.
Introduction Necrotizing sialometaplasia (NS) is an inflammatory disease of the salivary glands [1,2]. Its etiology is not scientifically proven, but it is thought to be the result of a physical or chemical aggression causing ischemia and subsequent infarction and necrosis of the gland, followed by repair [1,2]. The risk factors associated with this pathology include direct local trauma (ill-fitting dental prostheses, intubation, administering local anesthesia, local surgeries, violent vomiting, adjacent tumor) or systemic aggressions, which condition the reduction of perfusion (radiotherapy, tobacco,
∗
Corresponding author. Tel.: +351 966 493 711. E-mail address: [email protected] (T. Bernardo).
alcohol, diabetes) [1]. The sex ratio is preponderantly male (2:1), Caucasian, with an average age of 46. The presenting aspect is an ulcerated lesion with sharply defined borders, painful (in more than 50% of reported cases) and most often located on the hard palate although it may appear in any of the normal minor salivary gland locations, from the sinuses to the lungs [1—3]. Batsakis et al. have also described a case affecting the major salivary glands after surgery for an unrelated pathology [4]. The destruction of the hard palate bone is uncommon but does not exclude the diagnosis [2]. It is histologically characterized by necrosis of the glandular acini, conserving the lobular architecture associated with inflammation and squamous metaplasia of the ducts and the acini [1,2]. Other variably present findings include pseudoepitheliomatous hyperplasia or erosion of the epithelium and granulation tissue in the chorion. The proportion of the ischemic or metaplastic acini varies, and
1879-7296/$ – see front matter © 2012 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.anorl.2012.02.003
276
T. Bernardo et al.
that is why some authors consider five different histological stages (necrotic, sequestration, ulceration, reparative and healing) but it is uncommon to find them to be well differentiated in a biopsy [2]. Its evolution is self-limited in 4—10 weeks; therefore, the medical attitude after the biopsy confirming the diagnosis should be one of expectation [2]. Relapses are not frequent, although there are reported cases of repeat lesions [5,6]. The clinical importance of NS arises from the fact that its clinical and histological aspect may raise issues of differential diagnosis with malignant neoplasms namely, squamous cell and mucoepidermoid carcinoma, and eventually cystic adenoid carcinoma [1,2].
Case report A female patient, 43, Caucasian, came to the Hospital Emergency Service due to a painful 8-day-old neoformation on the palate that appeared in the postoperative period immediately after an abdominoplasty. She did not have any other complaints, namely systemic symptoms such as fever, anorexia or weight loss. She had been previously observed by a dentist who performed the puncture and the drainage of the lesion with release of the sero-hematic content and put her on antibiotics, with no improvement. As background she had morbid obesity, non-type-2 diabetes, gastric band surgery 4 years ago with a 40 kg weight loss and brachioplasty 2 years ago. She denied the overuse of alcoholic beverages or smoking. The physical examination showed an ulcerated and infiltrative lesion of the palate with sharply defined borders (Fig. 1). She did not have any palpable cervical adenopathies. A biopsy was performed and the anatomopathological result (Fig. 2) confirmed an NS. We kept up surveillance and after 6 weeks the lesion had spontaneously disappeared.
Figure 1
Lesion at the 8th day.
Discussion Our patient’s risk factor, prior to the onset of the lesion, was the trauma of orotracheal intubation performed to general anesthesia for abdominoplasty. The location of the lesion matches that which is described in the literature as the most frequent as well as its clinical presentation and self-resolution in 6 weeks. In the differential diagnosis of NS, the different neoplasms, aphthous lesions of the oral cavity, infectious processes such as tuberculosis and primary syphilis and acute necrotizing sialadenitis must not be forgotten [1]. This last entity is considered by many to be part of the NS spectrum (a subtype or an early phase) and is characterized by an acute unspecific inflammatory process with secondary acinar necrosis without ductal metaplasia or pseudoepitheliomatous hyperplasia [7]. The diagnosis of NS is based on the clinical and histological characteristics. The biopsy is paramount to avoid non-recommended aggressive treatments. In case of clinical need to clarify the lesion, the most appropriate imaging examination would be a MRI, whose main characteristics are the liquid aspect of the matter and the absence of contrast uptake [8]. Even histologically, this type of lesion can be hard to differentiate, essentially from: squamous cell and mucoepidermoid carcinoma. Another benign entity similar to the
Figure 2 Malpighian type mucosa with inflammatory polymorphic infiltrates, edema and vascular ectasis of the chorion.
aforementioned is the nicotinic stomatitis [9]. Conservation of lobular architecture of the metaplastic acini, in small amplification, is an important clue for diagnosing NS. In cases where the cytological atypia is marked, Rizkalla and Tonner [10] have investigated the use of myoepithelial markers (smooth muscle actin (SMA), calponin, p63), basal membrane markers (laminin and type IV collagen), E-cadherin and several cytokeratins (CK5, CK6, CK7, CAM5.2). They concluded that in NS they identified residual myoepithelial cells in the periphery of the epithelial islands that do not exist in the mucoepidermoid and squamous
Benign or malignant? Case report carcinoma. They also concluded that the low level of CK7 in NS can differentiate it from mucoepidermoid carcinoma, where level is much higher. In cases where the NS diagnosis is not clear, these panels of immunomarkers can be useful in the diagnosis, but are not pathognomonic. The analysis of several serial sections of well-targeted fragments, integrated in the clinical context, remains a better diagnostic approach [9]. It is important to monitor this disease until the full resolution of the lesion, since there may be an adjacent carcinoma that was accidentally left out of the biopsy material (there is a reported case of adenoid cystic carcinoma [3]). The tumor itself can be the cause of NS; therefore, coexistence is always a possibility. The ulcer heals spontaneously by secondary intention [2].
Conclusion The awareness of this pathological entity by the surgeon and the anatomopathologist is essential for the diagnosis. It is vital to histologically characterize all the ulcerated neoformations of the oral cavity so that the therapeutic orientation is the most appropriate and unsuitable approaches are avoided.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
277
References [1] Femopase FL, Hernández SL, Gendelman H, et al. Necrotizing sialometaplasia: report of five cases. Med Oral 2004;9: 304—8. [2] Imbery TA, Edwards PA. Necrotizing sialometaplasia: literature review and case reports. J Am Dent Assoc 1996;127: 1087—92. [3] Lee DJ, Ahn HK, Koh ES, et al. Necrotizing sialometaplasia accompanied by adenoid cystic carcinoma on the soft palate. Clin Exp Otorhinolaryngol 2009;2(1):48—51 [Epub 2009 Mar 26]. [4] Batsakis JG, Manning JT. Necrotizing sialometaplasia of major salivary glands. J Laryng Otol 1987;101:962—6. [5] Arguelles MT, Viloria JB, Talens MJ, et al. Necrotizing sialometaplasia. Oral Surg 1976;42:86—90. [6] Niedzielska I, Janic T, Markowski J. Bilateral localization of necrotizing sialometaplasia: a case report. Cases J 2009;2:9068. [7] Lombardi T, Samson J, Küffer R. Subacute necrotizing sialadenitis: a form of necrotizing sialometaplasia? Arch Otolaryngol Head Neck Surg 2003;129(9):972—5. [8] Farina D, Gavazzi E, Avigo C, et al. Case report. MRI findings of necrotizing sialometaplasia. Br J Radiol 2008;81(966): e173—5. [9] Carlson DL. Necrotazing sialometaplasia: a practical approach to the diagnosis. Arch Pathol Lab Med 2009;133(5):692—8. [10] Rizkalla H, Toner M. Necrotizing sialometaplasia versus invasive carcinoma of the head and neck: the use of myoepithelial markers and keratin subtypes as an adjunct to diagnosis. Histopathology 2007;51(2):184—9.
Available online at
www.sciencedirect.com
CASE REPORT
Benign or malignant? Case report T. Bernardo a,∗, D. Tente b, P. Oliveira a, D. Helena a, D. Ribeiro a, S. Gerós a, A. Silva a a
ORL Department, Centro Hospitalar de Vila Nova de Gaia/Espinho-EPE, Rua Conceic¸ão Fernandes, 4434-502 Vila Nova de Gaia, Portugal b Pathology Department, Centro Hospitalar de Vila Nova de Gaia/Espinho-EPE, Rua Conceic¸ão Fernandes, 4434-502 Vila Nova de Gaia, Portugal
KEYWORDS Necrotizing sialometoplasia; Salivary glands; Oral tumors
Summary Introduction: Necrotizing sialometoplasia (NS) is a disease in which the clinical and sometimes even histological features can be confused with those of a malignant tumor, but which is in fact a self-limited inflammatory lesion of the salivary glands. Case report: The authors present a case study of a 43-year-old female patient who came to the ENT Emergency Department with a painful ulcerated lesion of the hard palate. The lesion was 8 days old and had appeared after an abdominoplasty. Given the clinical need to rule out a malignant neoplasm, a biopsy was performed: the histological result was NS. Discussion/conclusion: The authors make a literature review and stress the importance of differentiating between NS and salivary gland neoplasia. Histology is essential in case of ulcerated neoformation of the oral cavity, to adapt treatment, avoiding unsuitable approaches. © 2012 Elsevier Masson SAS. All rights reserved.
Introduction Necrotizing sialometaplasia (NS) is an inflammatory disease of the salivary glands [1,2]. Its etiology is not scientifically proven, but it is thought to be the result of a physical or chemical aggression causing ischemia and subsequent infarction and necrosis of the gland, followed by repair [1,2]. The risk factors associated with this pathology include direct local trauma (ill-fitting dental prostheses, intubation, administering local anesthesia, local surgeries, violent vomiting, adjacent tumor) or systemic aggressions, which condition the reduction of perfusion (radiotherapy, tobacco,
∗
Corresponding author. Tel.: +351 966 493 711. E-mail address: [email protected] (T. Bernardo).
alcohol, diabetes) [1]. The sex ratio is preponderantly male (2:1), Caucasian, with an average age of 46. The presenting aspect is an ulcerated lesion with sharply defined borders, painful (in more than 50% of reported cases) and most often located on the hard palate although it may appear in any of the normal minor salivary gland locations, from the sinuses to the lungs [1—3]. Batsakis et al. have also described a case affecting the major salivary glands after surgery for an unrelated pathology [4]. The destruction of the hard palate bone is uncommon but does not exclude the diagnosis [2]. It is histologically characterized by necrosis of the glandular acini, conserving the lobular architecture associated with inflammation and squamous metaplasia of the ducts and the acini [1,2]. Other variably present findings include pseudoepitheliomatous hyperplasia or erosion of the epithelium and granulation tissue in the chorion. The proportion of the ischemic or metaplastic acini varies, and
1879-7296/$ – see front matter © 2012 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.anorl.2012.02.003
276
T. Bernardo et al.
that is why some authors consider five different histological stages (necrotic, sequestration, ulceration, reparative and healing) but it is uncommon to find them to be well differentiated in a biopsy [2]. Its evolution is self-limited in 4—10 weeks; therefore, the medical attitude after the biopsy confirming the diagnosis should be one of expectation [2]. Relapses are not frequent, although there are reported cases of repeat lesions [5,6]. The clinical importance of NS arises from the fact that its clinical and histological aspect may raise issues of differential diagnosis with malignant neoplasms namely, squamous cell and mucoepidermoid carcinoma, and eventually cystic adenoid carcinoma [1,2].
Case report A female patient, 43, Caucasian, came to the Hospital Emergency Service due to a painful 8-day-old neoformation on the palate that appeared in the postoperative period immediately after an abdominoplasty. She did not have any other complaints, namely systemic symptoms such as fever, anorexia or weight loss. She had been previously observed by a dentist who performed the puncture and the drainage of the lesion with release of the sero-hematic content and put her on antibiotics, with no improvement. As background she had morbid obesity, non-type-2 diabetes, gastric band surgery 4 years ago with a 40 kg weight loss and brachioplasty 2 years ago. She denied the overuse of alcoholic beverages or smoking. The physical examination showed an ulcerated and infiltrative lesion of the palate with sharply defined borders (Fig. 1). She did not have any palpable cervical adenopathies. A biopsy was performed and the anatomopathological result (Fig. 2) confirmed an NS. We kept up surveillance and after 6 weeks the lesion had spontaneously disappeared.
Figure 1
Lesion at the 8th day.
Discussion Our patient’s risk factor, prior to the onset of the lesion, was the trauma of orotracheal intubation performed to general anesthesia for abdominoplasty. The location of the lesion matches that which is described in the literature as the most frequent as well as its clinical presentation and self-resolution in 6 weeks. In the differential diagnosis of NS, the different neoplasms, aphthous lesions of the oral cavity, infectious processes such as tuberculosis and primary syphilis and acute necrotizing sialadenitis must not be forgotten [1]. This last entity is considered by many to be part of the NS spectrum (a subtype or an early phase) and is characterized by an acute unspecific inflammatory process with secondary acinar necrosis without ductal metaplasia or pseudoepitheliomatous hyperplasia [7]. The diagnosis of NS is based on the clinical and histological characteristics. The biopsy is paramount to avoid non-recommended aggressive treatments. In case of clinical need to clarify the lesion, the most appropriate imaging examination would be a MRI, whose main characteristics are the liquid aspect of the matter and the absence of contrast uptake [8]. Even histologically, this type of lesion can be hard to differentiate, essentially from: squamous cell and mucoepidermoid carcinoma. Another benign entity similar to the
Figure 2 Malpighian type mucosa with inflammatory polymorphic infiltrates, edema and vascular ectasis of the chorion.
aforementioned is the nicotinic stomatitis [9]. Conservation of lobular architecture of the metaplastic acini, in small amplification, is an important clue for diagnosing NS. In cases where the cytological atypia is marked, Rizkalla and Tonner [10] have investigated the use of myoepithelial markers (smooth muscle actin (SMA), calponin, p63), basal membrane markers (laminin and type IV collagen), E-cadherin and several cytokeratins (CK5, CK6, CK7, CAM5.2). They concluded that in NS they identified residual myoepithelial cells in the periphery of the epithelial islands that do not exist in the mucoepidermoid and squamous
Benign or malignant? Case report carcinoma. They also concluded that the low level of CK7 in NS can differentiate it from mucoepidermoid carcinoma, where level is much higher. In cases where the NS diagnosis is not clear, these panels of immunomarkers can be useful in the diagnosis, but are not pathognomonic. The analysis of several serial sections of well-targeted fragments, integrated in the clinical context, remains a better diagnostic approach [9]. It is important to monitor this disease until the full resolution of the lesion, since there may be an adjacent carcinoma that was accidentally left out of the biopsy material (there is a reported case of adenoid cystic carcinoma [3]). The tumor itself can be the cause of NS; therefore, coexistence is always a possibility. The ulcer heals spontaneously by secondary intention [2].
Conclusion The awareness of this pathological entity by the surgeon and the anatomopathologist is essential for the diagnosis. It is vital to histologically characterize all the ulcerated neoformations of the oral cavity so that the therapeutic orientation is the most appropriate and unsuitable approaches are avoided.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
277
References [1] Femopase FL, Hernández SL, Gendelman H, et al. Necrotizing sialometaplasia: report of five cases. Med Oral 2004;9: 304—8. [2] Imbery TA, Edwards PA. Necrotizing sialometaplasia: literature review and case reports. J Am Dent Assoc 1996;127: 1087—92. [3] Lee DJ, Ahn HK, Koh ES, et al. Necrotizing sialometaplasia accompanied by adenoid cystic carcinoma on the soft palate. Clin Exp Otorhinolaryngol 2009;2(1):48—51 [Epub 2009 Mar 26]. [4] Batsakis JG, Manning JT. Necrotizing sialometaplasia of major salivary glands. J Laryng Otol 1987;101:962—6. [5] Arguelles MT, Viloria JB, Talens MJ, et al. Necrotizing sialometaplasia. Oral Surg 1976;42:86—90. [6] Niedzielska I, Janic T, Markowski J. Bilateral localization of necrotizing sialometaplasia: a case report. Cases J 2009;2:9068. [7] Lombardi T, Samson J, Küffer R. Subacute necrotizing sialadenitis: a form of necrotizing sialometaplasia? Arch Otolaryngol Head Neck Surg 2003;129(9):972—5. [8] Farina D, Gavazzi E, Avigo C, et al. Case report. MRI findings of necrotizing sialometaplasia. Br J Radiol 2008;81(966): e173—5. [9] Carlson DL. Necrotazing sialometaplasia: a practical approach to the diagnosis. Arch Pathol Lab Med 2009;133(5):692—8. [10] Rizkalla H, Toner M. Necrotizing sialometaplasia versus invasive carcinoma of the head and neck: the use of myoepithelial markers and keratin subtypes as an adjunct to diagnosis. Histopathology 2007;51(2):184—9.