Squamous cell carcinoma of the tongue in a patient with Plummer-Vinson syndrome. Presentation of a case
tongue cancer causes /Why is tongue cancer produced? Tongue Cancer
Alejandro Donohue-Cornejo, * Dalia Abril Guzmán-Gastelum, * Daniel Constandse-Cortéz, * Luis Alberto Gaitán-Cepeda, § Carlos Reyes-Escalera§
* Department of Stomatology, Institute of Biomedical Sciences, Autonomous University of Ciudad Juárez.
§ Laboratory of Clinical and Experimental Pathology, Division of Postgraduate Studies and Research, Faculty of Dentistry, National Autonomous University of Mexico.
SUMMARY
Plummer-Vinson syndrome, also called sideropenic dysphagia, is characterized by dysphagia, the presence of esophageal membranes, and chronic iron-deficiency anemia, as well as an increased risk of developing carcinomas of the upper gastrointestinal tract. The reported cases of patients with Plummer-Vinson syndrome with carcinomas of the oral cavity are very rare. We present the case of a 45-year-old patient with a diagnosis of Plummer-Vinson syndrome who developed squamous cell carcinoma of the tongue. The patient presented at the Pathology and Oral Medicine Clinic of the Faculty of Dentistry of the Autonomous University of Ciudad Juárez for presenting dysphagia of 8 months of evolution, glosodynia and glossopyrosis. Her medical file revealed a history of unspecified chronic anemia, transfused several times in an inpatient manner. The intraoral examination showed depapilation of the anterior two thirds of the lingual dorsum, as well as an ulcerated volume increase of indurated edges, of unknown evolution in the left lateral border of the tongue. An incisional biopsy was performed with a histopathological diagnosis of squamous cell carcinoma. To our knowledge, this is the third case reported in the scientific literature of lingual squamous cell carcinoma in a patient suffering from Plummer-Vinson syndrome. The three cases are coincident in gender, age and depapilation of the back of the tongue. The relevance of continuing to include siderophenic dysphagia as a condition that favors the presence of intraoral carcinomas is discussed.
Key words: Plummer-Vinson syndrome, sideropenic dysphagia, squamous cell carcinoma, tongue.
INTRODUCTION
Plummer-Vinson syndrome is a rare syndrome characterized by dysphagia, the presence of esophageal membranes and chronic iron deficiency anemia. Its oral characteristics include glossitis, glossopyrosis, glossodynia and angular cheilitis.1 Its ethology is unknown, although autoimmune, genetic, infectious and nutritional factors have been proposed in its genesis.2
Approximately 10% of patients with Plummer-Vinson syndrome develop squamous cell carcinoma mainly in the hypopharynx and esophagus.3 The reported cases of patients with Plummer-Vinson syndrome who develop squamous cell carcinomas of the oral cavity are scarce, being even more rare cases of squamous cell carcinoma of the tongue.2 In order to increase the knowledge of the association between Plummer-Vinson syndrome and squamous cell carcinoma of the oral cavity, a case of a patient with a diagnosis of oral squamous cell carcinoma is presented. Plummer-Vinson who developed squamous cell carcinoma of the tongue.PRESENTATION OF THE CASE
In May 2004, a 45-year-old female patient presented to the Pathology and Oral Medicine Clinic of the Faculty of Dentistry of the Autonomous University of Ciudad Juárez for presenting dysphagia, glosodynia and glossopyrosis. Dysphagia is referred to approximately 8 months of evolution, increasing in the last six months. Glossodynia and glossopyrosis are constant and are exacerbated by hot and / or spicy foods. A history refers to a history of unspecified chronic anemia, transfused several times in an inpatient setting. It also refers to weight loss of unexplained origin greater than 10% of the total weight. Esophageal endoscopy was requested, which was performed with a private physician identifying the presence of esophageal membranes. Blood count values showed hematocrit and hemoglobin below normal with an important annocytosis and hypochromic microcytosis. The iron kinetic test corroborated deficiency, issuing a diagnosis of Plummer-Vinson syndrome, for which treatment with iron therapy was indicated.
The intraoral physical examination showed atrophy of the anterior two thirds of the lingual dorsum. An increase in volume was also identified, of unknown evolution in the left lateral border of the tongue that extended to the dorsal and ventral region of the tongue. Clinically, the lesion was composed of an ulcer with irregular borders, erythematous with whitish areas (Figure 1). Upon palpation the increase in volume was firm and painful under pressure. The patient denied a history of alcohol and smoking. With presumptive diagnosis of squamous cell carcinoma is referred to the Maxillofacial Surgery Service of the same institution for incisional biopsy. The surgical specimen was fixed in formalin buffered at 10%, to be embedded in paraffin and cut to 5 micrometers, stained with the hematoxylin and eosin technique. Microscopically the lesion was characterized by presenting neoplastic cells of epithelial origin with individual and group keratinizations; cellular and nuclear pleomorphism, nuclear hyperchromatism and increased and abnormal mitoses (Figure 2). The basement membrane was lost with infiltration of the neoplastic epithelium into connective and muscular tissue. A diagnosis of squamous cell carcinoma is issued.
The patient referred to a specialized cancer center where she underwent specialized treatment including surgery and radiotherapy. After five years he is in remission with periodic reviews.
DISCUSSION
It has been proposed that patients with Plummer-Vinson syndrome show a greater predisposition to develop squamous cell carcinoma, which is considered a pre-cancerous condition for the esophagus and stomach.4 It is associated with an increased incidence of post-cricoid squamous cell carcinoma. The association between this syndrome and the presence of solid-pseudopapillary carcinoma of the pancreas has also been suggested.5
Although the origin of the Plummer-Vinson syndrome is not known, it has been suggested that the main factor in its genesis is iron deficiency. The epithelium of the upper digestive tract is susceptible to iron deficiency, since its rapid rate of exchange causes it to rapidly lose iron-dependent enzymes.1 Thus, epithelial changes observed in patients with Plummer-Vinson syndrome can be due to a depletion of the oxidized enzymes of the epithelial cells. These changes produce atrophic glossitis, angular cheilitis, coilonychia, fragility and thinning of nails, fragility of the hair, and of special importance for the present article, promote the development of squamous cell carcinomas.6 However, in some cases the treatment with supplements iron is insufficient to reverse and improve patients with sidepenic dysphagia, so it is thought that there must be other factors, in addition to iron deficiency in the etiopathogenesis of this syndrome.1In our knowledge, only two cases of squamous cell carcinoma have been reported in the literature of patients diagnosed with Plummer-Vinson syndrome.7,8 In 1967, a case of a 52-year-old female patient with a deficiency was reported. iron that also had a depapilated area on the dorsal surface of the tongue, histopathologically confirming the diagnosis of squamous cell carcinoma.7 Later in 1970, Santoro et al8 reported a case of a 42-year-old female patient with dysphagia, anemia and asthenia, which clinically presented depapilation of the lingual dorsum and the presence of an ulcer with indurated borders. Histopathologically, the diagnosis of squamous cell carcinoma was also confirmed. The clinical description of the only two reported cases of squamous cell carcinoma in patients with Plummer-Vinson syndrome is, coinciding with the case presented here, this is a woman in the 5th decade of life who has a depapillated area on the back of the tongue and an ulcer with indurated edges.
Squamous cell carcinoma of the tongue has been associated with tobacco and alcohol consumption.9-11 The clinical file of the present case did not show any tobacco or alcoholic habit, which suggests association with the Plummer-Vinson syndrome. However, the fact that in the last decade has been reported the presence of squamous cell carcinoma of the oral cavity, specifically tongue, 11 in young subjects and without exposure to risk factors, raises the possibility that in the present case both entities have been developed synchronously.
It is noteworthy that Plummer-Vinson syndrome has traditionally been included as a risk condition for the development of carcinoma, especially of squamous cells of the oral cavity. The review of the literature shows few reports of cases of squamous cell carcinoma of the oral cavity in patients suffering from Plummer-Vinson syndrome. The most recent is a 2003 report of a patient with celiac disease and Plummer-Vinson syndrome who developed squamous cell carcinoma of the tongue.2 The only two reports found of patients with oral cavity carcinoma in subjects suffering from Plummer's syndrome Vinson have been published for more than 30 years. A possible justification for this fact is that the Plummer-Vinson syndrome is associated with social conditions of vulnerability, specifically poverty and malnutrition, so that improvements in socio-economic changes in different societies will result in a decrease in the incidence and prevalence of this syndrome. This scenario was predicted from the mid-703s of the last century and later confirmed in a Swedish population.12 However, in developing or underdeveloped countries where undernutrition may be a common event, the dental surgeon and especially the The doctor or oral pathologist should be alert to any suspicious lesion of malignancy, especially in women with malnutrition (anemia) who present with tongue depapilation.
Alejandro Donohue-Cornejo, * Dalia Abril Guzmán-Gastelum, * Daniel Constandse-Cortéz, * Luis Alberto Gaitán-Cepeda, § Carlos Reyes-Escalera§
* Department of Stomatology, Institute of Biomedical Sciences, Autonomous University of Ciudad Juárez.
§ Laboratory of Clinical and Experimental Pathology, Division of Postgraduate Studies and Research, Faculty of Dentistry, National Autonomous University of Mexico.
SUMMARY
Plummer-Vinson syndrome, also called sideropenic dysphagia, is characterized by dysphagia, the presence of esophageal membranes, and chronic iron-deficiency anemia, as well as an increased risk of developing carcinomas of the upper gastrointestinal tract. The reported cases of patients with Plummer-Vinson syndrome with carcinomas of the oral cavity are very rare. We present the case of a 45-year-old patient with a diagnosis of Plummer-Vinson syndrome who developed squamous cell carcinoma of the tongue. The patient presented at the Pathology and Oral Medicine Clinic of the Faculty of Dentistry of the Autonomous University of Ciudad Juárez for presenting dysphagia of 8 months of evolution, glosodynia and glossopyrosis. Her medical file revealed a history of unspecified chronic anemia, transfused several times in an inpatient manner. The intraoral examination showed depapilation of the anterior two thirds of the lingual dorsum, as well as an ulcerated volume increase of indurated edges, of unknown evolution in the left lateral border of the tongue. An incisional biopsy was performed with a histopathological diagnosis of squamous cell carcinoma. To our knowledge, this is the third case reported in the scientific literature of lingual squamous cell carcinoma in a patient suffering from Plummer-Vinson syndrome. The three cases are coincident in gender, age and depapilation of the back of the tongue. The relevance of continuing to include siderophenic dysphagia as a condition that favors the presence of intraoral carcinomas is discussed.
Key words: Plummer-Vinson syndrome, sideropenic dysphagia, squamous cell carcinoma, tongue.
INTRODUCTION
Plummer-Vinson syndrome is a rare syndrome characterized by dysphagia, the presence of esophageal membranes and chronic iron deficiency anemia. Its oral characteristics include glossitis, glossopyrosis, glossodynia and angular cheilitis.1 Its ethology is unknown, although autoimmune, genetic, infectious and nutritional factors have been proposed in its genesis.2
Approximately 10% of patients with Plummer-Vinson syndrome develop squamous cell carcinoma mainly in the hypopharynx and esophagus.3 The reported cases of patients with Plummer-Vinson syndrome who develop squamous cell carcinomas of the oral cavity are scarce, being even more rare cases of squamous cell carcinoma of the tongue.2 In order to increase the knowledge of the association between Plummer-Vinson syndrome and squamous cell carcinoma of the oral cavity, a case of a patient with a diagnosis of oral squamous cell carcinoma is presented. Plummer-Vinson who developed squamous cell carcinoma of the tongue.PRESENTATION OF THE CASE
In May 2004, a 45-year-old female patient presented to the Pathology and Oral Medicine Clinic of the Faculty of Dentistry of the Autonomous University of Ciudad Juárez for presenting dysphagia, glosodynia and glossopyrosis. Dysphagia is referred to approximately 8 months of evolution, increasing in the last six months. Glossodynia and glossopyrosis are constant and are exacerbated by hot and / or spicy foods. A history refers to a history of unspecified chronic anemia, transfused several times in an inpatient setting. It also refers to weight loss of unexplained origin greater than 10% of the total weight. Esophageal endoscopy was requested, which was performed with a private physician identifying the presence of esophageal membranes. Blood count values showed hematocrit and hemoglobin below normal with an important annocytosis and hypochromic microcytosis. The iron kinetic test corroborated deficiency, issuing a diagnosis of Plummer-Vinson syndrome, for which treatment with iron therapy was indicated.
The intraoral physical examination showed atrophy of the anterior two thirds of the lingual dorsum. An increase in volume was also identified, of unknown evolution in the left lateral border of the tongue that extended to the dorsal and ventral region of the tongue. Clinically, the lesion was composed of an ulcer with irregular borders, erythematous with whitish areas (Figure 1). Upon palpation the increase in volume was firm and painful under pressure. The patient denied a history of alcohol and smoking. With presumptive diagnosis of squamous cell carcinoma is referred to the Maxillofacial Surgery Service of the same institution for incisional biopsy. The surgical specimen was fixed in formalin buffered at 10%, to be embedded in paraffin and cut to 5 micrometers, stained with the hematoxylin and eosin technique. Microscopically the lesion was characterized by presenting neoplastic cells of epithelial origin with individual and group keratinizations; cellular and nuclear pleomorphism, nuclear hyperchromatism and increased and abnormal mitoses (Figure 2). The basement membrane was lost with infiltration of the neoplastic epithelium into connective and muscular tissue. A diagnosis of squamous cell carcinoma is issued.
The patient referred to a specialized cancer center where she underwent specialized treatment including surgery and radiotherapy. After five years he is in remission with periodic reviews.
DISCUSSION
It has been proposed that patients with Plummer-Vinson syndrome show a greater predisposition to develop squamous cell carcinoma, which is considered a pre-cancerous condition for the esophagus and stomach.4 It is associated with an increased incidence of post-cricoid squamous cell carcinoma. The association between this syndrome and the presence of solid-pseudopapillary carcinoma of the pancreas has also been suggested.5
Although the origin of the Plummer-Vinson syndrome is not known, it has been suggested that the main factor in its genesis is iron deficiency. The epithelium of the upper digestive tract is susceptible to iron deficiency, since its rapid rate of exchange causes it to rapidly lose iron-dependent enzymes.1 Thus, epithelial changes observed in patients with Plummer-Vinson syndrome can be due to a depletion of the oxidized enzymes of the epithelial cells. These changes produce atrophic glossitis, angular cheilitis, coilonychia, fragility and thinning of nails, fragility of the hair, and of special importance for the present article, promote the development of squamous cell carcinomas.6 However, in some cases the treatment with supplements iron is insufficient to reverse and improve patients with sidepenic dysphagia, so it is thought that there must be other factors, in addition to iron deficiency in the etiopathogenesis of this syndrome.1In our knowledge, only two cases of squamous cell carcinoma have been reported in the literature of patients diagnosed with Plummer-Vinson syndrome.7,8 In 1967, a case of a 52-year-old female patient with a deficiency was reported. iron that also had a depapilated area on the dorsal surface of the tongue, histopathologically confirming the diagnosis of squamous cell carcinoma.7 Later in 1970, Santoro et al8 reported a case of a 42-year-old female patient with dysphagia, anemia and asthenia, which clinically presented depapilation of the lingual dorsum and the presence of an ulcer with indurated borders. Histopathologically, the diagnosis of squamous cell carcinoma was also confirmed. The clinical description of the only two reported cases of squamous cell carcinoma in patients with Plummer-Vinson syndrome is, coinciding with the case presented here, this is a woman in the 5th decade of life who has a depapillated area on the back of the tongue and an ulcer with indurated edges.
Squamous cell carcinoma of the tongue has been associated with tobacco and alcohol consumption.9-11 The clinical file of the present case did not show any tobacco or alcoholic habit, which suggests association with the Plummer-Vinson syndrome. However, the fact that in the last decade has been reported the presence of squamous cell carcinoma of the oral cavity, specifically tongue, 11 in young subjects and without exposure to risk factors, raises the possibility that in the present case both entities have been developed synchronously.
It is noteworthy that Plummer-Vinson syndrome has traditionally been included as a risk condition for the development of carcinoma, especially of squamous cells of the oral cavity. The review of the literature shows few reports of cases of squamous cell carcinoma of the oral cavity in patients suffering from Plummer-Vinson syndrome. The most recent is a 2003 report of a patient with celiac disease and Plummer-Vinson syndrome who developed squamous cell carcinoma of the tongue.2 The only two reports found of patients with oral cavity carcinoma in subjects suffering from Plummer's syndrome Vinson have been published for more than 30 years. A possible justification for this fact is that the Plummer-Vinson syndrome is associated with social conditions of vulnerability, specifically poverty and malnutrition, so that improvements in socio-economic changes in different societies will result in a decrease in the incidence and prevalence of this syndrome. This scenario was predicted from the mid-703s of the last century and later confirmed in a Swedish population.12 However, in developing or underdeveloped countries where undernutrition may be a common event, the dental surgeon and especially the The doctor or oral pathologist should be alert to any suspicious lesion of malignancy, especially in women with malnutrition (anemia) who present with tongue depapilation.
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