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An unusual cause of otalgia in a child – A case report
pediatria polska 91 (2016) 480–483
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevier.com/locate/pepo
Case report/Kazuistyka
An unusual cause of otalgia in a child – A case report Santosh Kumar Swain 1,*, Mahesh Chandra Sahu 2, Kabikanta Samantray 3 1
Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan University, Odisha, India Directorate of Medical Research, IMS and SUM Hospital, Siksha “O” Anusandhan University, Odisha, India 3 Department of Otorhinolaryngology, KIMS, Bhubaneswar, Odisha, India 2
article info
abstract
Article history:
Otalgia is a common complaint of pediatric patients in Otorhinolaryngological practice.
Received: 08.05.2016
Rhinogenic contact point otalgia (RCPO) is a newer term in medicine. This is due to
Accepted: 17.05.2016
intranasal mucosal contact points which formed in nasal septal deviation, septal spur,
Available online: 26.05.2016
concha bullosa, etc. causing referred Otalgia without any signs of inflammation. It is diagnosed after exclusion of other causes for ear pain after different investigation tools.
Keywords: Otalgia Rhinogenic contact point otalgia Rhinogenic contact point headache
Here, we reported a case 12-year boy presented with intractable otalgia induced by nasal septal spur. Otoendoscopy revealed normal tympanic membrane whereas diagnostic nasal endoscopy showed a sharp spur touching the left inferior turbinate in left nostril. Medical treatment for migraine failed to relieve the symptoms. After surgical removal of septal spur, patient experienced a significant relief of symptoms. © 2016 Polish Pediatric Society. Published by Elsevier Sp. z o.o. All rights reserved.
Introduction Otalgia is a common clinical entity of childhood in Otolaryngological practice and is nearly universal in the course of everyone's life. It is a distressing problem for child as well as parents. Pressure of two opposing mucosa in the nasal cavity without evidence of inflammation can be a cause of headache or facial pain, called as rhinogenic contact point headache. Rhinogenic contact point headache (RCPH) is a referred pain due to intranasal contact between the nasal septum and lateral nasal wall. Headache and facial pain can occur due to intranasal mucosal contact points such as septal deviation, septal spur, large ethmoidal bulla and
concha bullosa of middle turbinate. Intranasal contact points denotes to a contact between two opposing intranasal mucosal surfaces. Intranasal contact points are present in about 4% of noses [1]. If there are no features of inflammation in sinonasal area nor other etiology for headache, it should be in mind to assess the intranasal contact point headache. Otalgia due to rhinogenic contact point by intranasal mucosa is called as rhinogenic contact point Otalgia (RCPO) [2]. Diagnosis of RCPO needs a team approach. In our case, a 12 year old boy who presented with intermittent and severe otalgia, which was supposed to be induced by sharp septal spur touching to the inferior turbinate. After surgical correction of nasal septal spur, the otalgia has been alleviated. Nose has a diverse anatomical variation like septal spur, septal deviations, concha bullosa
* Corresponding author at: Department of Otorhinolaryngology, IMS and SUM Hospital, Kalinga Nagar, Bhubaneswar 3, Odisha, India. Tel.: +91 9556524887. E-mail address: [email protected] (S.K. Swain). http://dx.doi.org/10.1016/j.pepo.2016.05.005 0031-3939/© 2016 Polish Pediatric Society. Published by Elsevier Sp. z o.o. All rights reserved.
pediatria polska 91 (2016) 480–483
Fig. 1 – Otoendoscopy showing normal left tympanic membrane
481
Fig. 2 – Diagnostic nasal endoscopy showing a sharp spur in left nostril touching to the inferior turbinate
and large ethmoidal bulla, etc. Relation between these anatomical variations and rhinogenic contact point otalgia was confirmed in septal spur. So above lesions should not be ignored from mind during evaluation of otalgia and their respective treatment helps to relief the symptoms.
Case report A 12-year-old boy attended outpatient Department of Otorhinolaryngology with complaints of severe left side ear pain since 2 years. The characteristic ear pain or otalgia was deep and sharp shooting in character. The otalgia was intermittent and occasionally lasts for several hours. The pain often affecting left ear and sometimes radiating to the left side forehead area. He had no history of cold and fever. He was treated for migraine at outside hospital, but not relieved. He showed normal neurological and ophthalmological examinations. The otoendoscopic examination revealed bilateral normal external auditory canal and tympanic membrane (Fig. 1). Diagnostic nasal endoscopy was done by using 08 rigid nasal endoscope which showed a sharp spur from left side nasal septum touching to the inferior turbinate (Fig. 2). During diagnostic nasal endoscopy, there was no purulent discharge and no sign of inflammation seen. Computed tomography (CT) scan confirmed a sharp spur in left side of septum which has a distinct contact point between the nasal septum and inferior turbinate (Fig. 3). 4% xylocaine with adrenaline soaked cotton pledget was kept at mucosal contact point of septal spur and after around 3 min patient felt an improvement of otalgia. Then patient was advised for removal of mucosal contact point by spurectomy/septoplasty. Patient completely recovered after 2 weeks of
Fig. 3 – CT scan of the paranasal sinus showing a sharp spur in left nostril touching to the left inferior turbinate
482
pediatria polska 91 (2016) 480–483
surgery. Intranasal mucosal contact point should be considered as an etiological factor for causing referred otalgia.
Discussion Otalgia is a common complaint in medicine. There may be primary cause confined to the lesion in ear or secondary to lesions at other site, called as referred otalgia. The intranasal mucosal contact point relating to the otalgia, called as rhinogenic contact point otalgia (RCPO) is new terminology in medicine. Wolf in 1942 first documented the concept of referred headache secondary to intranasal mucosal contact points and described that facial pain and headache can occur due to contact between the turbinates and other parts of nasal cavity [1]. This type of headache is called as rhinogenic contact point headache (RCPH) which is a referred pain over face and head, arising from contact point between the mucosa of nasal septum and lateral wall of the nose [3]. The headache in RCPH is referred through the trigeminal nerve [4]. The cause of RCPH is multifactorial. RCPH may result from nociceptors in the nasal mucosa, which ends up in the sensory nucleus of the trigeminal nerve. Our case was showing no evidence of inflammatory pathology in nose and sinus except a sharp spur causing intranasal mucosal contact point. He has no refractory error and normal neurological examination. Pressure effect on the nasal mucosa is associated with changes in microvascular supply, followed by release of biologic substances, induces pain or decreasing the pain threshold. The contact between mucosal lining of concha bullosa and nasal septum or the lateral wall of nose result in release of substance P, calcitonin gene related peptide (CGRP) [5] and neurokinin A [6]. These chemicals are found in nociceptive fibers in the central nervous system and trigeminovascular system. So the contact point between intranasal mucosa may be a cause of secondary headache or triggering factor to primary headache [7]. This phenomenon is also called as middle turbinate syndrome [8]. Substance P (SP) has a known role in pathophysiology of contact point headache. Substance P is a neuropeptide that can be identified in the mucosa of the nasal cavity. When SP is released around vascular area, vasodilatation, plasma extravasations and perivascular inflammation occurs, causing headache similar to clinical manifestations of migraine without aura [1]. Normal nasal mucosa has a higher concentration of SP than chronic hyperplastic mucosa or polypoidal tissue. This explains why contact point headache are almost always seen in patients without rhinosinusitis. RCPH is frequently seen in septal deviations/spur followed by concha bullosa of middle turbinates. Creating a space between middle turbinate and septum is needed for reversing this situation. This is done by trimming the parts of middle turbinate. Exact mechanism for different characteristic pain in various anatomical variations of nose is not known. It is thought that large contact point as in lamella bullosa and tight contact as in sharp spur may cause severe contact point headache. Diagnostic nasal endoscopy in conjunction with CT scan has proven to be ideal combination for diagnosis of sinonasal pathology. Anatomical variations like septal deviation, spurs, concha bullosa, hypertrophied inferior turbinate, medialized middle turbinate,
uncinate bulla, medially or laterally bent uncinate process, paradoxically middle turbinate, large ethmoidal bulla are often cause for headache. In our case, a 12 year boy was complaining intermittent left side otalgia along with radiating pain to left side forehead. Patient had frequently visited to pediatrician, neurologist and ophthalmologist where all clinical examinations showed normal findings. By proper and precise investigations with the help of rigid nasal endoscopy and CT scan of paranasal sinus and brain, we confirmed the spur in left nostril touching to the inferior turbinate and there was no evidence of inflammation in nose and sinus. This type of otalgia is called as rhinogenic contact point otalgia by few author. So, always careful evaluations of anatomical variations of the nose should be done with the help of diagnostic nasal endoscopy and computed tomography (CT) scan in chronic headache. These anatomical variations should treated surgically. After evolution of endoscopic sinus surgery, techniques like partial turbinectomy, turbinoplasty, spurectomy or septoplasy are practiced aiming to relieve the contact point headaches [5].
Conclusion Otalgia is a symptom with which Otolaryngologist and pediatrician deal frequently. Rhinogenic contact point otalgia is rarely documented in medicine. Otalgia without any clear etiology, the intranasal mucosal contact points must be evaluated. CT scan and diagnostic nasal endoscopy are the best methods for evaluation of these factors to find out rhinogenic cause. Otalgia due to contact point of intranasal mucosa may be considered as exclusion of diagnosis and it should be included in the differential diagnosis for etiology of otalgia. This presentation gives awareness to the pediatrician and Otolaryngologist regarding intranasal mucosal contact point which may be a cause of referred otalgia.
Authors’ contributions/Wkład autorów According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie None declared.
Ethics/Etyka The work described in this article has been carried out in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal
pediatria polska 91 (2016) 480–483
experiments; Uniform Requirements for manuscripts submitted to Biomedical journals.
r e f e r e n c e s / p i s m i e n n i c t w o
[1] Peric A, Baletic N, Sotirovic J. A case of an uncommon anatomic variation of the middle turbinate associated with headache. Acta Otorhinolaryngol Ital 2010;30:156–159. [2] Kim SH. A case of nasal septal deviation-induced rhinogenic contact point otalgia. Am J otolaryngol 2015;36:451–455. [3] Roozbahany NA, Nasri S. Nasal and paranasal sinus anatomical variations in patients with rhinogenic contact point headache. Auris Nasus Larynx 2013;40:177–183.
483
[4] Behin F, Behin B, Bigal ME, Lipton RB. Surgical treatment of patients with refractory migraine headaches and intranasal contact points. Cephalalgia 2005;25:439–443. [5] Durham PL. Calcitonin gene-related peptide (CGRP) and migraine. Headache 2006;46(Suppl. 1):3–8. [6] Goadsby PJ, Hoskin KL, Storer RJ, Edvinsson L, Connor HE. Adenosine A1 receptor agonists inhibit trigeminovascular nociceptive transmission. Brain 2002;125 (Pt 6):1392–1401. [7] Behin F, Lipton RB, Bigal M. Migraine and intranasal contact point headache: is there any connection? Curr Pain Headache Rep 2006;10(4):312–315. [8] Anselmo-Lima WT, De Oliveira JA, Speciali JG, Bordini C, Dos Santos AC, Rocha KV, et al. Middle turbinate headache syndrome. Headache 1997;37:102–106.
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevier.com/locate/pepo
Case report/Kazuistyka
An unusual cause of otalgia in a child – A case report Santosh Kumar Swain 1,*, Mahesh Chandra Sahu 2, Kabikanta Samantray 3 1
Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan University, Odisha, India Directorate of Medical Research, IMS and SUM Hospital, Siksha “O” Anusandhan University, Odisha, India 3 Department of Otorhinolaryngology, KIMS, Bhubaneswar, Odisha, India 2
article info
abstract
Article history:
Otalgia is a common complaint of pediatric patients in Otorhinolaryngological practice.
Received: 08.05.2016
Rhinogenic contact point otalgia (RCPO) is a newer term in medicine. This is due to
Accepted: 17.05.2016
intranasal mucosal contact points which formed in nasal septal deviation, septal spur,
Available online: 26.05.2016
concha bullosa, etc. causing referred Otalgia without any signs of inflammation. It is diagnosed after exclusion of other causes for ear pain after different investigation tools.
Keywords: Otalgia Rhinogenic contact point otalgia Rhinogenic contact point headache
Here, we reported a case 12-year boy presented with intractable otalgia induced by nasal septal spur. Otoendoscopy revealed normal tympanic membrane whereas diagnostic nasal endoscopy showed a sharp spur touching the left inferior turbinate in left nostril. Medical treatment for migraine failed to relieve the symptoms. After surgical removal of septal spur, patient experienced a significant relief of symptoms. © 2016 Polish Pediatric Society. Published by Elsevier Sp. z o.o. All rights reserved.
Introduction Otalgia is a common clinical entity of childhood in Otolaryngological practice and is nearly universal in the course of everyone's life. It is a distressing problem for child as well as parents. Pressure of two opposing mucosa in the nasal cavity without evidence of inflammation can be a cause of headache or facial pain, called as rhinogenic contact point headache. Rhinogenic contact point headache (RCPH) is a referred pain due to intranasal contact between the nasal septum and lateral nasal wall. Headache and facial pain can occur due to intranasal mucosal contact points such as septal deviation, septal spur, large ethmoidal bulla and
concha bullosa of middle turbinate. Intranasal contact points denotes to a contact between two opposing intranasal mucosal surfaces. Intranasal contact points are present in about 4% of noses [1]. If there are no features of inflammation in sinonasal area nor other etiology for headache, it should be in mind to assess the intranasal contact point headache. Otalgia due to rhinogenic contact point by intranasal mucosa is called as rhinogenic contact point Otalgia (RCPO) [2]. Diagnosis of RCPO needs a team approach. In our case, a 12 year old boy who presented with intermittent and severe otalgia, which was supposed to be induced by sharp septal spur touching to the inferior turbinate. After surgical correction of nasal septal spur, the otalgia has been alleviated. Nose has a diverse anatomical variation like septal spur, septal deviations, concha bullosa
* Corresponding author at: Department of Otorhinolaryngology, IMS and SUM Hospital, Kalinga Nagar, Bhubaneswar 3, Odisha, India. Tel.: +91 9556524887. E-mail address: [email protected] (S.K. Swain). http://dx.doi.org/10.1016/j.pepo.2016.05.005 0031-3939/© 2016 Polish Pediatric Society. Published by Elsevier Sp. z o.o. All rights reserved.
pediatria polska 91 (2016) 480–483
Fig. 1 – Otoendoscopy showing normal left tympanic membrane
481
Fig. 2 – Diagnostic nasal endoscopy showing a sharp spur in left nostril touching to the inferior turbinate
and large ethmoidal bulla, etc. Relation between these anatomical variations and rhinogenic contact point otalgia was confirmed in septal spur. So above lesions should not be ignored from mind during evaluation of otalgia and their respective treatment helps to relief the symptoms.
Case report A 12-year-old boy attended outpatient Department of Otorhinolaryngology with complaints of severe left side ear pain since 2 years. The characteristic ear pain or otalgia was deep and sharp shooting in character. The otalgia was intermittent and occasionally lasts for several hours. The pain often affecting left ear and sometimes radiating to the left side forehead area. He had no history of cold and fever. He was treated for migraine at outside hospital, but not relieved. He showed normal neurological and ophthalmological examinations. The otoendoscopic examination revealed bilateral normal external auditory canal and tympanic membrane (Fig. 1). Diagnostic nasal endoscopy was done by using 08 rigid nasal endoscope which showed a sharp spur from left side nasal septum touching to the inferior turbinate (Fig. 2). During diagnostic nasal endoscopy, there was no purulent discharge and no sign of inflammation seen. Computed tomography (CT) scan confirmed a sharp spur in left side of septum which has a distinct contact point between the nasal septum and inferior turbinate (Fig. 3). 4% xylocaine with adrenaline soaked cotton pledget was kept at mucosal contact point of septal spur and after around 3 min patient felt an improvement of otalgia. Then patient was advised for removal of mucosal contact point by spurectomy/septoplasty. Patient completely recovered after 2 weeks of
Fig. 3 – CT scan of the paranasal sinus showing a sharp spur in left nostril touching to the left inferior turbinate
482
pediatria polska 91 (2016) 480–483
surgery. Intranasal mucosal contact point should be considered as an etiological factor for causing referred otalgia.
Discussion Otalgia is a common complaint in medicine. There may be primary cause confined to the lesion in ear or secondary to lesions at other site, called as referred otalgia. The intranasal mucosal contact point relating to the otalgia, called as rhinogenic contact point otalgia (RCPO) is new terminology in medicine. Wolf in 1942 first documented the concept of referred headache secondary to intranasal mucosal contact points and described that facial pain and headache can occur due to contact between the turbinates and other parts of nasal cavity [1]. This type of headache is called as rhinogenic contact point headache (RCPH) which is a referred pain over face and head, arising from contact point between the mucosa of nasal septum and lateral wall of the nose [3]. The headache in RCPH is referred through the trigeminal nerve [4]. The cause of RCPH is multifactorial. RCPH may result from nociceptors in the nasal mucosa, which ends up in the sensory nucleus of the trigeminal nerve. Our case was showing no evidence of inflammatory pathology in nose and sinus except a sharp spur causing intranasal mucosal contact point. He has no refractory error and normal neurological examination. Pressure effect on the nasal mucosa is associated with changes in microvascular supply, followed by release of biologic substances, induces pain or decreasing the pain threshold. The contact between mucosal lining of concha bullosa and nasal septum or the lateral wall of nose result in release of substance P, calcitonin gene related peptide (CGRP) [5] and neurokinin A [6]. These chemicals are found in nociceptive fibers in the central nervous system and trigeminovascular system. So the contact point between intranasal mucosa may be a cause of secondary headache or triggering factor to primary headache [7]. This phenomenon is also called as middle turbinate syndrome [8]. Substance P (SP) has a known role in pathophysiology of contact point headache. Substance P is a neuropeptide that can be identified in the mucosa of the nasal cavity. When SP is released around vascular area, vasodilatation, plasma extravasations and perivascular inflammation occurs, causing headache similar to clinical manifestations of migraine without aura [1]. Normal nasal mucosa has a higher concentration of SP than chronic hyperplastic mucosa or polypoidal tissue. This explains why contact point headache are almost always seen in patients without rhinosinusitis. RCPH is frequently seen in septal deviations/spur followed by concha bullosa of middle turbinates. Creating a space between middle turbinate and septum is needed for reversing this situation. This is done by trimming the parts of middle turbinate. Exact mechanism for different characteristic pain in various anatomical variations of nose is not known. It is thought that large contact point as in lamella bullosa and tight contact as in sharp spur may cause severe contact point headache. Diagnostic nasal endoscopy in conjunction with CT scan has proven to be ideal combination for diagnosis of sinonasal pathology. Anatomical variations like septal deviation, spurs, concha bullosa, hypertrophied inferior turbinate, medialized middle turbinate,
uncinate bulla, medially or laterally bent uncinate process, paradoxically middle turbinate, large ethmoidal bulla are often cause for headache. In our case, a 12 year boy was complaining intermittent left side otalgia along with radiating pain to left side forehead. Patient had frequently visited to pediatrician, neurologist and ophthalmologist where all clinical examinations showed normal findings. By proper and precise investigations with the help of rigid nasal endoscopy and CT scan of paranasal sinus and brain, we confirmed the spur in left nostril touching to the inferior turbinate and there was no evidence of inflammation in nose and sinus. This type of otalgia is called as rhinogenic contact point otalgia by few author. So, always careful evaluations of anatomical variations of the nose should be done with the help of diagnostic nasal endoscopy and computed tomography (CT) scan in chronic headache. These anatomical variations should treated surgically. After evolution of endoscopic sinus surgery, techniques like partial turbinectomy, turbinoplasty, spurectomy or septoplasy are practiced aiming to relieve the contact point headaches [5].
Conclusion Otalgia is a symptom with which Otolaryngologist and pediatrician deal frequently. Rhinogenic contact point otalgia is rarely documented in medicine. Otalgia without any clear etiology, the intranasal mucosal contact points must be evaluated. CT scan and diagnostic nasal endoscopy are the best methods for evaluation of these factors to find out rhinogenic cause. Otalgia due to contact point of intranasal mucosa may be considered as exclusion of diagnosis and it should be included in the differential diagnosis for etiology of otalgia. This presentation gives awareness to the pediatrician and Otolaryngologist regarding intranasal mucosal contact point which may be a cause of referred otalgia.
Authors’ contributions/Wkład autorów According to order.
Conflict of interest/Konflikt interesu None declared.
Financial support/Finansowanie None declared.
Ethics/Etyka The work described in this article has been carried out in accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal
pediatria polska 91 (2016) 480–483
experiments; Uniform Requirements for manuscripts submitted to Biomedical journals.
r e f e r e n c e s / p i s m i e n n i c t w o
[1] Peric A, Baletic N, Sotirovic J. A case of an uncommon anatomic variation of the middle turbinate associated with headache. Acta Otorhinolaryngol Ital 2010;30:156–159. [2] Kim SH. A case of nasal septal deviation-induced rhinogenic contact point otalgia. Am J otolaryngol 2015;36:451–455. [3] Roozbahany NA, Nasri S. Nasal and paranasal sinus anatomical variations in patients with rhinogenic contact point headache. Auris Nasus Larynx 2013;40:177–183.
483
[4] Behin F, Behin B, Bigal ME, Lipton RB. Surgical treatment of patients with refractory migraine headaches and intranasal contact points. Cephalalgia 2005;25:439–443. [5] Durham PL. Calcitonin gene-related peptide (CGRP) and migraine. Headache 2006;46(Suppl. 1):3–8. [6] Goadsby PJ, Hoskin KL, Storer RJ, Edvinsson L, Connor HE. Adenosine A1 receptor agonists inhibit trigeminovascular nociceptive transmission. Brain 2002;125 (Pt 6):1392–1401. [7] Behin F, Lipton RB, Bigal M. Migraine and intranasal contact point headache: is there any connection? Curr Pain Headache Rep 2006;10(4):312–315. [8] Anselmo-Lima WT, De Oliveira JA, Speciali JG, Bordini C, Dos Santos AC, Rocha KV, et al. Middle turbinate headache syndrome. Headache 1997;37:102–106.