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Rhinotillexomania in a Cystic Fibrosis Patient Resulting in Septal Perforation Mark Gelpi1*, Emily N Ahadizadeh1,2, Brian D’Anzaa1 and Kenneth Rodriguez1

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Rhinotillexomania in a Cystic Fibrosis Patient Resulting in Septal Perforation Mark Gelpi1*, Emily N Ahadizadeh1,2, Brian D’Anzaa1 and Kenneth Rodriguez1 ISSN: 2572-4193 Gelpi et al. J Otolaryngol Rhinol 2018, 4:036 DOI: 10.23937/2572-4193.1510036 Volume 4 | Issue 1 Journal of Open Access Otolaryngology and Rhinology CASE REPORT Rhinotillexomania in a Cystic Fibrosis Patient Resulting in Septal Perforation Mark Gelpi1*, Emily N Ahadizadeh1,2, Brian D’Anzaa1 and Kenneth Rodriguez1 1 Check for University Hospitals Cleveland Medical Center, USA updates 2Case Western Reserve University School of Medicine, USA *Corresponding author: Mark Gelpi, MD, University Hospitals Cleveland Medical Center, 11100 Euclid Avenue, Cleveland, OH 44106, USA, Tel: (216)-844-8433, Fax: (216)-201-4479, E-mail: [email protected] paranasal sinuses [1,4]. Nasal symptoms in CF patients Abstract occur early, manifesting between 5-14 years of age, and Cystic fibrosis (CF) is a multisystem disease that can have represent a life-long problem in this population [5]. Pa- significant sinonasal manifestations. Viscous secretions are one of several factors in CF that result in chronic sinona- tients with CF can develop thick nasal secretions con- sal pathology, such as sinusitis, polyposis, congestion, and tributing to chronic rhinosinusitis (CRS), nasal conges- obstructive crusting. Persistent discomfort and nasal man- tion, nasal polyposis, headaches, and hyposmia [6-8]. ifestations of this disease significantly affect quality of life. Sinonasal symptoms of CF are managed medically with Digital manipulation and removal of crusting by the patient in an attempt to alleviate the discomfort can have unfore- topical agents and antibiotics, however surgery can be seen damaging consequences. We present one such case warranted due to the chronic and refractory nature of and investigate other cases of septal damage secondary to the symptoms, with 20-25% of CF patients undergoing digital trauma, as well as discuss the importance of sinona- sinus surgery in their lifetime [8]. sal therapy in CF patients. Although sinus involvement is widespread in CF pa- Keywords tients, one complication that, to our knowledge, has Septal perforation, Cystic fibrosis, Digital trauma, Rhino- not been reported as a phenomenon is septal perfo- tillexomania, Nose picking ration from rhinotillexomania. Equally uncommon are reports of septal perforation in patients secondary to Introduction digital trauma (nose-picking). There have been a hand- Cystic fibrosis (CF) is a systemic, inherited disease ful of reports of septal destruction resulting from ha- caused by mutations in the cystic fibrosis transmem- bitual, aggressive digital trauma also known as rhino- brane conductance regulator gene that results in im- tillexomania (compulsive nose picking). These cases paired anion transport, affecting the respiratory, exo- have been reported in the context of chronic nasal dis- crine, gastrointestinal, and reproductive tracts [1]. The comfort or psychiatric disease [9-11]. Septal perfora- impairment of ion transport results in impaired muco- tion is traditionally associated with intranasal cocaine ciliary clearance and characteristically viscous secre- abuse, systemic rheumatologic disease, granulomatosis with polyangiitis, and glucocorticoid induced atrophy, tions, which acts as a nidus for inflammation, infection, among other causes [12-18]. Since this defect is com- and edema [2]. monly a manifestation of systemic disease, any patient Involvement of the respiratory tract is a major cause presenting with idiopathic septal perforation merits of morbidity in this population, and upper airway in- thorough investigation into the etiology. We report a volvement occurs in 90-100% of cases [3]. This disease case of a 32-year-old woman with a history of CF pre- can have profound effects on the nasal mucosa and senting with facial pain and pressure whom was found Citation: Gelpi M, Ahadizadeh EN, D’Anzaa B, Rodriguez K (2018) Rhinotillexomania in a Cystic Fi- brosis Patient Resulting in Septal Perforation. J Otolaryngol Rhinol 4:036. doi.org/10.23937/2572- 4193.1510036 Received: March 01, 2018: Accepted: April 28, 2018: Published: April 30, 2018 Copyright: © 2018 Gelpi M, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Gelpi et al. J Otolaryngol Rhinol 2018, 4:036 • Page 1 of 4 • DOI: 10.23937/2572-4193.1510036 ISSN: 2572-4193 Figure 1: Computed tomography of septal perforation measuring 21.6 mm in the cranial to caudal direction and 27.4 mm in the anterior to posterior direction. to have obliteration of the nasal septum, extensive tur- ally without active polyposis or purulence. CT imaging binate damage, lamina papyracea erosion, and partial showed sinuses consistent with left medial antrectomy, ethmoidectomy secondary to digital trauma. partial left ethmoidectomy, and bilateral middle turbi- nate resection, without evidence of polyposis, although Case Description this patient reported that she had never undergone si- A 32-year-old woman with CF and a past medical nus surgery. The septal perforation on the CT scan mea- history of seizures and depression presented to the rhi- sured 21.6 mm in height and 27.4 mm in the anterior to nology clinic with facial pain and pressure. She was a posterior direction (Figure 1). former smoker who had been hospitalized three times On further questioning, the patient stated that she in the last ten years for respiratory exacerbations of used her fingers to chronically manipulate and pick her her chronic disease. Her CF was diagnosed at thirteen nose. She noted that after a period of time of digital ma- months of age by sweat chloride test (results of 102 and nipulation, she was able to insert her finger on one side 108) and was confirmed by genetic testing (N1303K/ of her nose and manipulate the other side. She report- F508del variant). She is managed at University Hospi- ed persistent irritation of her nose and manipulated it tals Pulmonary Cystic Fibrosis Clinic. Her lung function in order to obtain relief as well as to clear secretions at presentation demonstrated a FVC of 3.01 L, a FEV / 1 and crusting. It was concluded that the changes seen on FVC ratio of 59%, and an FEV of 51% of predicted value, 1 endoscopy and imaging were secondary to digital ma- consistent with severe disease. On initial presentation, nipulation. Additionally, a diagnosis of CRS was made she reported an extensive history of sinus discomfort, and the patient received a course of levofloxacin. The atopic disease, daily migraines, and recent fluctuations patient’s condition did not improve and she later under- in her sense of taste and smell, as well as sleep distur- went surgical intervention with bilateral frontal sinuso- bance, fatigue, throat clearing, itchy eyes, and epistaxis. tomies, total ethmoidectomies, maxillary antorstomies Her only pulmonary symptom at that time was short- with tissue removal, and sphenoidotomies with de- ness of breath. The patient was using oral antihista- bridement of inflamed tissue and mucosa. The patholo- mines, fluticasone spray, and routine saline irrigation gy report for the tissue obtained at the time of surgery to manage her symptoms, and had recently received a showed evidence of chronic inflammation and did not course of oral steroids and clarithromycin from an out- show evidence of any rheumatologic disease process. side provider for acute sinusitis. Additionally, she was on tiotropium and albuterol aerosols, tobramycin neb- Conclusion ulizer, a multivitamin, and enzyme replacement thera- To our knowledge, there have been no documented py including Pulmozyne and Pertzye. She denied nasal reports of manual destruction of the nasal septum and drainage, nasal airway obstruction, dysphonia, sneez- turbinates in CF patients, or an increased prevalence of ing, or aspirin sensitivity. septal perforation in this population. Additionally, there Sinonasal endoscopy was performed and demon- are very few documented cases of extensive septal and strated a large septal perforation as well as the absence turbinate destruction resulting from digital trauma, as of the anterior aspect of the middle turbinate bilater- was seen in this patient [9-11,19]. Gelpi et al. J Otolaryngol Rhinol 2018, 4:036 • Page 2 of 4 • DOI: 10.23937/2572-4193.1510036 ISSN: 2572-4193 The degree of self-inflicted damage seen in this case management. In addition, in the broader population, it reflects the lengths to which patients will go in order to is important to keep rhinotillexomania on the differen- relieve their discomfort and highlights a need for symp- tial when evaluating a patient with septal perforation of tom management in patients with CF and nasal crust- unknown etiology. ing. Conservative management with aerosolized nasal steroids, antihistamines, and saline irrigation has been Conflict of Interest regarded as the initial step in treating chronic sinona- There is no conflict of interest. sal complaints in CF patients [20]. However, guidelines for dosing and interval of treatment have not been out- Funding Disclosures lined, which may result in inappropriate and ineffica- None declared. cious therapies. The use of extended duration empiric antibiotic therapy, both topical and systemic, for these References symptoms is particularly concerning, considering the 1. Clunes MT, Boucher RC (2007) Cystic fibrosis: The mech- high risk that CF patients have of colonization with mul- anisms of pathogenesis of an inherited lung disorder. Drug
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