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CASE REPORT
Year : 2022 | Volume
: 8
| Issue : 4 | Page : 217-220
Esophageal melanosis due to mishri usage: A case with uncommon finding and rare etiology
Muppa Indrakeela Girish, Amol Sonyabapu Dahale, Debabrata Banerjee, Prasad Bhate, Abhijeet Karad
Department of Gastroenterology and Hepatology, Dr. D.Y Patil Medical College, Hospital and Research Centre, Dr. D.Y Patil Vidyapeeth, Pune, Maharashtra, India
Correspondence Address:
Dr. Amol Sonyabapu Dahale
Department of Gastroenterology and Hepatology, Dr. D.Y Patil Medical College, Hospital and Research Centre, Dr. D.Y Patil Vidyapeeth, Pune, Maharashtra
India
Source of Support: None, Conflict of Interest: None
CheckDOI: 10.4103/ijam.ijam_67_22
Esophageal melanosis (EM) is a rare disease of the upper digestive system. We present a case series of two patients with EM who presented with dysphagia and had the habit of mishri consumption. Endoscopy showed black-pigmented spots involving the upper and mid esophagus. EM due to mishri consumption was a rare presentation as there were no cases reported till date.
The following core competencies are addressed in this article: Patient care, Practice-based learning and improvement, Medical knowledge.
Keywords: Dysphagia, esophageal melanosis, esophagogastroduodenoscopy mishri
Esophageal melanosis (EM) is a very rare disease of the digestive system. It was first described by De la Pava in 1963, and its prevalence in autopsies was reported to be 4%.[1] The esophagus lacks melanocytes. During early embryogenesis, melanocytes normally migrate from the neural crest to other organs, likely the epidermis and oral cavity. Abnormal migration of melanocytes during embryogenesis takes place and basal melanocytes have been found in a small number of the normal esophagus.[2],[3] The exact reason for melanosis was unclear, but the hypothesis was chronic inflammatory stimuli and GERD, which damage the mucosa.[4] Some studies reported melanosis is a benign condition and considered it a precursor to carcinoma and melanoma, but there was no firm link proven. As EM is very rare, we report a case series of EM with unusual presentation.
CasesCase 1
A 60-year-old woman presented with a history of vomiting and dysphagia for solids for the past 3 months. No history was suggestive of reflux symptoms and weight loss. A history of smokeless tobacco (mishri) consumption is present. A history of comorbidities such as diabetes and hypertension (HTN) was present.
The physical examination and initial blood tests were normal. The chest X-ray and ultrasound abdomen were normal. Esophagogastroduodenoscopy was performed using the Fujifilm EG-760R, and a black-pigmented circular spot was noted at the upper and mid esophagus [Figure 1], from which multiple biopsies were taken from both the upper and mid lesions and showed erythema in the peripyloric region. A rapid urease test was done, which showed negative. Histopathology of the esophageal biopsies showed squamous mucosa with normal differentiation with no evidence of dysplasia. A striking feature is the presence of benign melanocytes scattered along the basal layer with intracytoplasmic dusty brown melanin pigment [Figure 2]. The patient was started on a proton pump inhibitor and was advised to follow up after 6 months for esophagogastroduodenoscopy.
Figure 1: Endoscopic view showing black-pigmented macular spots seen at upper and mid esophagus (blue arrow)
Figure 2: High power microscopy on hematoxylin and eosin stain (magnifying power 20x) shows melanocytes with dusty brown pigment (red arrow)Case 2
A 50-year-old woman presented with a 2–3-month history of dysphagia for solids and liquids, vomiting, and generalized weakness. There was no history of abdominal pain or reflux symptoms or weight loss. A history of smokeless tobacco (mishri) consumption is present. History of hypertension was present.
The physical examination and initial blood tests were normal.
The chest X-ray and abdominal ultrasound were both normal. Contrast-enhanced computerized tomography of the abdomen and pelvis was normal. Esophagogastroduodenoscopy was performed using the Fujifilm EG-760R, and a black-pigmented spot was noted at the upper and mid esophagus, from which multiple biopsies were taken from both the upper and mid lesions and showed erythematous mucosa at the antrum. The rapid urease test was negative. Histopathology of the esophageal biopsies showed esophageal squamous mucosa with increased melanin pigment in the basal layer [Figure 3]. These cells are positive for the HMB-45 stain [Figure 4]. The lamina propria shows melanin pigment with mild chronic inflammation. The patient was started on a proton pump inhibitor and was advised to follow up after 6 months for esophagogastroduodenoscopy.
Figure 3: On hematoxylin and eosin stain (magnifying power 20x) shows brown pigment in basal layer (red arrow)
Figure 4: Immunohistochemical stain (magnifying power 20x) for HMB 45 shows positive (green arrow) Discussion EM is a rare benign condition that is characterized by the proliferation of melanocytes in the basal layer of the esophagus and the accumulation of melanin pigment in the mucosa of the esophagus with a male preponderance.[2] In our case series, the mean age was 55 years. EM is more commonly seen in men. In Sharma et al.[5] study, males were more common than females (16:5). In a few case reports, most of the patients were females in their 5th decade. In most of the case reports, the patients' complaints are dyspeptic symptoms and rarely dysphagia. In the current case series, both patients are presented with dysphagia. On endoscopy, EM is commonly seen in the mid and lower esophagus and is described as flat, irregular black-pigmented spots. In Sharma et al.[5] study, the endoscopic features commonly seen in the mid-esophagus are followed by the lower and upper esophagus. In Kozan and Tatar[6] study, the endoscopic features were seen in the lower esophagus. The endoscopic features are described as flat, circular, oval, and linear pigmented black spots. In the present case series, the endoscopic features described as flat, black-pigmented spots are seen in the mid-esophagus. Histologically, EM is characterized by the presence of increased numbers of melanocytes in the basal layer of esophageal squamous epithelium and an increased quantity of melanin in the esophageal mucosa. On H and E staining, the melanocytes are pigment-laden dendritic cells. These cells are positive for melanocytic markers, such as S100, Melan-A, and HMB-45. In our study, histology shows melanocytes with dusty brown pigment in the basal layer, and these cells are positive for the HMB-45 stain.
Tobacco use leads to chronic damage to the upper gastrointestinal mucosa. In the current case series, both patients had a history of smokeless tobacco (mishri) consumption. The differential diagnosis of EM included benign melanotic nevi, blue naevi, and primary malignant melanoma because melanin deposition is the main feature in these lesions.[7] Some authors like Yokoyama A suggest EM as a precursor of malignant melanoma and carcinoma. In Ohnuma et al.[8] case series, the study described primary malignant melanoma of the esophagus is often accompanied by melanocytosis which was very difficult to differentiate because of their similar appearance. In Kinugasa et al.[9] study, they reported a case of EM with eosinophilic esophagitis but we have not found EM with eosinophilic esophagitis in our study. Unverdı et al.[10] described EM as perhaps the precursor lesion in EM, so they recommend clinical and endoscopic follow-up. In our case series, we started proton pump inhibitors and advised follow-up after 6 months to relook the endoscopy for any malignant findings. Therefore, EM patients required follow-up at frequent intervals to look for any malignancy.
ConclusionEM is a rare benign disorder. In our case series, there was a history of mishri consumption. EM due to mishri consumption was a rare presentation as there were no cases reported till date. A few case reports suggest that melanosis acts as a precursor for malignant transformation. Neither treatment nor surveillance is currently available. Follow-up with endoscopies is compulsory to observe for malignant transformation.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Research Quality and Ethics Statement
The authors of this manuscript declare that this scientific work complies with reporting quality, formatting, and reproducibility guidelines set forth by the EQUATOR Network. The authors also attest that this clinical investigation was determined to not require the Institutional Review Board/Ethics Committee review, and the corresponding protocol/approval number is not applicable. The authors certify that they have obtained appropriate patient consent. The patients have given their consent for their clinical information to be reported in the journal. The patients understand that their names and initials will not be published, and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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