Monthly bleeding nasal endometriosis: a case report

A 32-year-old nulliparous woman presented with cyclic nasal bleeding and fullness since puberty. No history of other site bleeding. She has no self or family history of diabetes mellitus or hypertension. No family history of similar compliant. Upon examination, vital signs were within normal range. Nasal endoscopic examination showed polypoid mass with smooth outer surface. Nasopharynx was normal. On laboratory workup, the white blood cell count was 9.6 × 10 9 L, with neutrophils of 66% and lymphocytes of 34%. Hemoglobin was 12.8 g/dl, and the platelet count was 234 × 10 9 / L. Blood group & Rh was A +. Fasting blood sugar, blood urea nitrogen, creatinine, liver function tests, and serum electrolytes were all within the normal range. Head and neck CT revealed a polypoid mass with no connection with the brain or meninges. Polypectomy was decided with the impression of a benign nasal polyp. Then, excision was done and submitted for biopsy. The lesional tissue (Fig. 1 ) was fixed in 10% neutral-buffered formalin, serial sectioned done, and stained with hematoxylin and eosin. Figure 1. Gross picture of the polypoid mass. Gross picture of the polypoid mass. The microscopy revealed stratified respiratory epithelium-lined polyploid tissue with submucous variably sized convoluted secretory glands lined by single layer of columnar cells with abundant apical cytoplasm and intraluminal content. Glands are embedded in dense, edematous stroma. Accompanying thick-walled arterioles were also noted (Figs 2 and 3 ). Figure 2. Low-power view (4×) – polypoid tissue lined by respiratory epithelium (white arrow) composed of subepithelial endometrial glands (black arrow) and stroma (blue arrow). Figure 3. Mid-power view (20×) – tissue lined by respiratory epithelium (white arrow) composed of subepithelial endometrial glands (black arrow) and stroma (blue arrow). Low-power view (4×) – polypoid tissue lined by respiratory epithelium (white arrow) composed of subepithelial endometrial glands (black arrow) and stroma (blue arrow). Mid-power view (20×) – tissue lined by respiratory epithelium (white arrow) composed of subepithelial endometrial glands (black arrow) and stroma (blue arrow). After polypectomy follow-up, for consecutive three months patient was event-free.

Endometriosis is a chronic gynecological disease defined with the presence of normal endometrial stroma and glands other than the uterine cavity [ 1 – 3 ] . It is relatively common condition, with peak occurrence in between 30 and 45 years [ 2 ] . Depending on the position of implantation, endometriosis is classified as extrapelvic or endopelvic. Although extrapelvic endometriosis is uncommon, many organs are involved by endometriosis including the gastrointestinal tract, urinary tract, upper and lower respiratory system, diaphragm, pleura, and pericardium, as well as abdominal scar sites [ 2 ] . The most frequent locations for extrapelvic endometriosis are the gastrointestinal and urinary tracts [ 1 ] . Nasal septal mucosal localized endometriosis is extremely rare [ 1 , 4 , 5 ] ; there are only two cases in English literature [ 2 ] . Both types of endometrioses can be diagnosed with well-defined histopathologic criteria [ 5 ] . We present an extremely rare nasal localized endometriosis case report to contribute to the literature. Physicians should be aware that nasal septal endometriosis may infrequently cause nasal lesions, nasal pain, nasal fullness, and recurrent cyclic perimenstrual epistaxis. This case report is written following the SCARE criteria [ 6 ] . HIGHLIGHTS Extrapelvic endometriosis is uncommon, and many organs are involved by endometriosis including the gastrointestinal tract, urinary tract, upper and lower respiratory system, diaphragm, pleura, and pericardium, as well as abdominal scar sites. Nasal septal mucosal localized endometriosis is very rare. Our case is a 32-year-old female patient who has presented with cyclic nasal bleeding and fullness. Histopathologic examination confirms the diagnosis of nasal endometriosis. HIGHLIGHTS Extrapelvic endometriosis is uncommon, and many organs are involved by endometriosis including the gastrointestinal tract, urinary tract, upper and lower respiratory system, diaphragm, pleura, and pericardium, as well as abdominal scar sites. Nasal septal mucosal localized endometriosis is very rare. Our case is a 32-year-old female patient who has presented with cyclic nasal bleeding and fullness. Histopathologic examination confirms the diagnosis of nasal endometriosis.

Endometriosis is an estrogen-dependent disease defined by the presence of normal endometrial mucosa abnormally implanted in locations other than the uterine cavity [ 2 ] . It is considered to be the most common cause of chronic pelvic pain in reproductive age since it is highly estrogen dependent. Although it is quit uncommon, it can occur in postmenopause [ 7 ] . Depending on the anatomical location involved by endometriosis, patients may manifest with hematuria, hematemesis, and epistaxis in urinary bladder, intestine, and upper respiratory, tract respectively [ 2 , 8 ] . This patient presented with cyclic nasal bleeding. Endometriosis affects 10–15% of females most commonly in reproductive age group with a peak age of 30–45 years [ 7 ] . There are many theories about the etiopathogenesis of endometriosis and the cause of intrapelvic and extrapelvic localizations [ 9 ] . Traditionally, there are three theories that attempt to explain the mechanism and cause of endometriosis [ 7 ] . The first theory states on the metaplasia of the peritoneal serosa cuff. According to the theory, endometriosis occurs in clusters in different tissues due to the effect of inflammatory or hormonal factors as a result of tissue differentiation of mesothelial cells of the peritoneum [ 10 ] . The second theory states that retrograde menstrual blood reflux and endometriotic cells are implanted in tissues in the peritoneal cavity. This can explain the intrapelvic localization of endometriosis but not the extrapelvic location [ 10 ] . The third theory is the intra- and extrapelvic spread of endometrial cells, just like cancer cells, through hematogenous and lymphogenic [ 11 ] . Lymphatic or hematogenous spread, genetics, and other modern theories are trying to explain how nasal endometriosis occurs as distant localization, but it is unclear yet [ 12 ] . However, extrapelvic nasal endometriosis should certainly be considered in females of reproductive age who have concurrent nasal bleeding with the menstrual cycle, nasal pain, the feeling of fullness, and nasal lesions [ 13 ] . The main diagnostic methods for diagnosis of endometriosis are as follows: ultrasonography – from the available imaging techniques, ultrasound has been proven useful in the diagnosis of endometriosis. Magnetic resonance imaging – in controversial cases, it can confirm the diagnosis of endometriosis and rule out other diseases. Computed tomography (CT) – it is a fast and readily accessible imaging technique. The examination is well tolerated and therefore suited to very elderly or infirm patients as well as children, people with claustrophobia, or patients who are critically ill. A CT scan shows the anatomical variations and the extent of the disease and is essential if surgical treatment is to be implemented. It should not be regarded as the primary step in the diagnosis of the condition, except where there are unilateral signs and symptoms or other sinister features, but rather corroborates history and endoscopic findings after failure of medical therapy. Intravenous contrast medium injection is not required unless a tumor, vascular lesion, or acute complication is suspected. Severe polyposis and unilateral opacification of the sinuses is accessed with a contrast-enhanced study to help distinguish mucosa, polyps, and fluid, and rule out any additional underlying pathology [ 14 ] . Diagnostic laparoscopy using biopsy tissue is still the most accurate way to diagnose problems. For this patient, diagnostic rhinoscopy with biopsy was done. The only way to diagnose endometriosis is by laparoscopy or other types of surgery including biopsy of the lesion. The diagnosis is based on the characteristic appearance of the disease and should be confirmed by biopsy. The surgery also allows for diagnosis of the surgical treatment of endometriosis at the same time [ 15 , 16 ] . In relation to histopathologic diagnosis whether it is pelvic or extrapelvic endometriosis, there are certain criteria and at least two of them must be fulfilled for the definitive diagnosis [ 7 ] . In this case, all the three criteria were fulfilled and final diagnosis was nasal endometriosis. Immunohistochemistry can be done rarely in uncertain cases for histopathology. since glandular structures express cytokeratin 7, estrogen, and progesterone, they can be used as a marker. In cases of stromal endometriosis, CD 10 is used as a preferred marker [ 9 ] . Management of endometriosis includes both medical and surgical options, determined by the organ involved, the patient’s response to medical treatment, and the desire to preserve reproductive capacity. The medical management includes contraceptive pills, which usually cause reduction in amount of bleeding and reduction in pain [ 7 ] . Surgical management could be conservative or non-conservative. Conservative surgical management includes electrofulguration, laser evaporation, or thermal coagulation. Surgical management is advised if medical management fails to control the symptoms [ 7 ] . For our patient, medical managements were not tried since diagnostic excisional biopsy was done and the patient showed complete resolutions of symptoms upon follow-up.

Nasal endometriosis is a very rare clinical entity, and it should certainly be considered in females of reproductive age who have concurrent nasal bleeding with the menstrual cycle, nasal pain, the feeling of fullness, and nasal lesions. Clinical information along with diagnostic laparoscopy and biopsy is used to confirm the diagnosis.