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Nasal Mucosa Ulceration and Septal Perforation As Initial Presentation of a Patient

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Nasal Mucosa Ulceration and Septal Perforation As Initial Presentation of a Patient International Journal of Pediatric Otorhinolaryngology Extra 9 (2014) 56–59 Contents lists available at ScienceDirect International Journal of Pediatric Otorhinolaryngology Extra jo urnal homepage: www.elsevier.com/locate/ijporl Case Report Nasal mucosa ulceration and septal perforation as initial presentation of a patient with probable Behcet’s disease Annika Meyer, Mary Czerny, Jastin Antisdel * Department of Otolaryngology, Saint Louis University School of Medicine, United States A R T I C L E I N F O A B S T R A C T Article history: Behcet’s disease is a vasculitis that affects multiple systems in the body, classically oral and genital Received 3 September 2013 mucosa. Nasal mucosa involvement is a rare manifestation of the disease, reported in only a handful of Accepted 7 January 2014 patients. We present an HLA-B51 positive, suspected Behcet’s patient with predominantly nasal manifestations including epistaxis, nasal mucosa ulceration and septal perforation. Her nasal symptoms Keywords: have improved since being started on a daily regimen of colchicine. We believe this case supports the Behcet’s disease wide clinical heterogeneity of the disorder, and serves as a reminder to consider Behcet’s disease when a Behcet syndrome patient presents with an atypical head and neck manifestation. HLA-B5 ß 2014 Elsevier Ireland Ltd. All rights reserved. HLA-B51 Nasal septum perforation Nasal ulcer 1. Report of a case room and showed evidence of ulceration, acute and chronic inflammation, squamous metaplasia, and fibrosis. A 14-year-old girl presented to an academic pediatric Nasal irrigation and topical antibiotics were prescribed. Over otolaryngology clinic with a 2-year history of recurrent epistaxis. the next three months the nasal ulcer decreased in size by She described the episodes as typical nosebleeds, usually from the approximately 40% by endoscopic visualization, though she was right nostril; they would last for about 10 min and could be still having regular epistaxis. She also complained of chronic stopped with pressure. No further intervention had been neces- headaches and recurrent small oral ulcers. The patient’s other sary. Rigid nasal endoscopy performed bilaterally revealed an medical problems include nodular acne vulgaris, hyperhidrosis of inflamed, abnormal-appearing mucosa with an ulcerated lesion the axillae, and attention deficit hyperactivity disorder, for which over her right septum covered with a thin crust (Figs. 1 and 2). The she is receiving treatment with norgestimate-ethinyl estradiol, etiology of the lesion was unclear. There was no history of trauma, axillary botulinum injections, and a combination of aripiprazole illicit drug abuse, or application of nasal sprays. The patient and methylphenidate, respectively. Her acne vulgaris has been appeared to be Caucasian with fair skin, but information on her resistant to oral contraceptive treatment, with failure to improve ethnic background was not available as she was adopted. on two prior formulations. She has also been noted to have Granulomatosis with polyangiitis (GPA, formerly Wegner’s gran- persistent livedo reticularis on physical exam. ulomatosis), Churg Strauss syndrome, and sarcoidosis were The following month she began to bleed from the left nostril. considered but C-ANCA, P-ANCA, and glomerular basement She was found to have a small ulceration and a hypervascular area membrane antibodies were negative, and an angiotensin convert- on the left septum (Fig. 3), which was cauterized and packed with ing enzyme (ACE) level was normal. A chest radiograph showed no Gelfoam. This lesion was biopsied and found to have pathology evidence of hilar adenopathy. The patient denied symptoms very similar to the previous biopsy. She continued to have near- suggestive of other sinonasal, lung, or kidney problems. A daily epistaxis. Lab results at this time showed elevated total laboratory evaluation including ESR, LDH, CK, TSH, and T4 was complement, with normal levels of C3 and C4. She had normal, and anti-thyroglobulin and anti-TPO antibodies were indeterminate test results for lupus anticoagulant. Her IgM and negative. A septal mucosal biopsy was performed in the operating IgG anticardiolipin, histone, and beta-2 glycoprotein antibodies were negative. The remaining autoimmune workup was negative, including rheumatoid factor, ANA, chromatin, dsDNA, SS-A, SS-B, * Corresponding author. Tel.: +1 314 577 8884; fax: +1 314 268 5111. E-mail address: [email protected] (J. Antisdel). as well as Smith, RNP, and cyclic citrullin peptide antibodies. 1871-4048/$ – see front matter ß 2014 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.pedex.2014.01.003 A. Meyer et al. / International Journal of Pediatric Otorhinolaryngology Extra 9 (2014) 56–59 57 Fig. 1. Right nasal lesion. Endoscopic view of the crusted, ulcerated lesion of the Fig. 3. Left nasal lesion. Endoscopic view of small ulceration and more anterior mucosa of the right nasal septum. hypervascular region of the left nasal septum. Several weeks later, the patient presented to the emergency gynecologic exam showed normal vaginal mucosa with no room with epistaxis. The ulceration had eroded through the nasal ulceration. She has, however, complained of persistent joint pain, septum, resulting in a septal perforation. A septal button was burning with urination, and recurrent right eye blurriness. Given placed at that time (Fig. 4). The patient was referred to positivity for HLA-B51 in the setting of oral and nasal ulceration rheumatology for further evaluation. Her Factor V Leiden, and myriad nonspecific (but potentially rheumatologic) com- prothrombin, antithrombin III, protein C and protein S were plaints, the patient was started on colchicine 0.6 mg daily for normal. She was found to have a heterozygous A1298C mutation presumed Behcet’s disease. Her epistaxis has decreased from 3–4 for methylenetetrahydrofolate reductase (MTHFR). Anti-Saccha- to 1–2 episodes per week since starting the colchicine. She romyces cerevisiae antibodies IgG and IgA were negative. She continues to have a septal perforation with septal button in place was found to be a carrier of HLA-B51, which is found in greater and is followed by rheumatology. than 60% of patients with Behcet’s disease. She was noted to develop a red bump at the needle stick site immediately 2. Discussion following lab draws, but true pathergy should occur 24–48 h later. An ophthalmologic exam was negative for uveitis and a Behcet’s disease is a systemic leukocytoclastic vasculitis characterized by recurrent episodes of mucosal apthae, classically of the oral and genital mucosa, and inflammatory ocular involvement such as uveitis. However, any system may be affected, with multiple other manifestations involving the skin, joints, vessels, as well as the CNS and gastrointestinal tract [1]. Involvement of the nasal mucosa in Behcet’s disease is rare and Fig. 2. Right nasal lesion, another view. Intraoperative endoscopic view of the lesion on the right nasal septum after crust removal. Fig. 4. Septal button. Endoscopic view of the septal button from the left nasal cavity. 58 A. Meyer et al. / International Journal of Pediatric Otorhinolaryngology Extra 9 (2014) 56–59 only a few cases have been reported in the literature. In a cross- Other explanations for the contributory relationship include sectional study of nasal involvement in Behcet’s patients, only 2 cross-reactivity between self-antigens and HLA-B51 itself [9], or out of 400 patients reviewed had nasal ulcers [2]. More common the presence of a linkage disequilibrium with a nearby and truly examples of nasal involvement included dysosmia in 15 patients causal gene such as the nearby alleles for tumor necrosis factor and nasal obstruction in 10 patients. No patients reported a history and MHC class I-related (MIC-A and MIC-B) genes, which are of epistaxis or septal perforation. involved in the immune response [9,10]. However, the contribu- Perforations of the nasal septum occur when the blood supply tion of HLA-B51 to the overall genetic susceptibility is estimated to the avascular cartilage is interrupted bilaterally at the same to be only 20%, implying that genes outside the MHC complex are location. The most common cause is iatrogenic injury from prior involved in the pathogenesis of Behcet’s disease [10]. Recently, nasal surgery, followed by trauma, nasal spray use, inhaled drug two large genome-wide association studies reported a correlation use, autoimmune disease, neoplasia, and inflammatory or infec- between single nucleotide polymorphisms (SNP) of interleukin tious origins [3]. A literature search revealed only one published (IL)-10 and IL-23R/IL-12RB2 genes and Behcet’s disease [10]. case report of a Behcet’s patient presenting with a destructive Evidently the pathogenesis of Behcet’s disease is complicated, and sinusopathy, including septal perforation and amputation of without a clear etiology it has been difficult to delineate effective conchae [4]. That patient had severe widespread disease with pharmacotherapy. middle ear involvement and hearing loss, dysphonic laryngitis, The mainstays of treatment of Behcet’s disease rely on severe oral and genital ulcerations, and skin manifestations. The immunosuppression and anti-inflammation. Topical and systemic currently presented case is the first to our knowledge of a patient corticosteroids may be used to control the inflammatory process. with suspected Behcet’s disease presenting predominantly with Many immunosuppressant agents are used, including
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