Home About us Editorial board Ahead of print Current issue Archives Submit article Instructions Subscribe Search Contacts Login 
  • Users Online: 340
  • Home
  • Print this page
  • Email this page


 
 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 29  |  Issue : 2  |  Page : 129-131

A rare case of Charlin's syndrome


Department of Oral Medicine and Radiology, Pacific Dental College, Debari, Rajasthan, India

Date of Submission28-Aug-2016
Date of Acceptance25-Oct-2017
Date of Web Publication9-Nov-2017

Correspondence Address:
Aveek Mukherji
Department of Oral Medicine and Radiology, Pacific Dental College, Debari, Rajasthan
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.JIAOMR_104_16

Rights and Permissions
   Abstract 

Charlin's syndrome is an extremely rare condition characterized by pain in the nasal and paranasal areas, which is precipitated by touching the lateral aspect of the ipsilateral nostril. We are presenting one such case of a 42-year-old man who was admitted to Dr. Chaudhary Hospital and Medical Research Centre.

Keywords: Charlin's syndrome, nasal nerve syndrome, nasociliaris nerve syndrome, nasociliary syndrome, nasociliary neuralgia


How to cite this article:
Singh MP, Mukherji A, Vats AK. A rare case of Charlin's syndrome. J Indian Acad Oral Med Radiol 2017;29:129-31

How to cite this URL:
Singh MP, Mukherji A, Vats AK. A rare case of Charlin's syndrome. J Indian Acad Oral Med Radiol [serial online] 2017 [cited 2018 Aug 18];29:129-31. Available from: http://www.jiaomr.in/text.asp?2017/29/2/129/217904


   Introduction Top


Charlin's syndrome is a very rare pathological condition, resulting from neuralgia of the nasociliary nerve.[1] It is also called nasal nerve syndrome, nasociliaris nerve syndrome, and nasociliary syndrome.[2] Some authors have referred to it as nasociliary neuralgia.[3] In this syndrome, touching the outer aspect of one nostril causes a lancinating pain,[4] which mainly radiates to the eyebrow, orbit, or into the nose or jaw, and is also accompanied by lacrimation, conjunctival infection, nasal congestion, sneezing, and redness of the skin on the forehead.[5] Here, one such case is being presented. A brief discussion about the diagnostic criteria and differential diagnosis has been provided, which emphasises on the ideal steps for management of such a condition.


   Case Report Top


A 42-year-old male patient presented to Dr. Chaudhary Hospital and Medical Research Centre with the chief complaint of pain in his nose and left eye since the last 5–6 days. The patient gave a history of a sharp lancinating pain in the left lateral nostril and the left eye since 5–6 days. The pain was sudden in onset, high in intensity, intermittent, aggravated on touching the nose and left eye, and relieved after 10–45 min without any medications. There were approximately 5 to 10 such episodes daily. The pain extended to the forehead and the left eye and was associated with a watery nasal discharge and tears in the left eye. There was no past history of such a condition. Patient had noticed some ulcerations over the left side of the nose after beginning of the pain, which gradually dried with time. Patient's appetite, bowel, micturition, and sleep were normal. There was no history of weight loss, fever, or cough. Patient's family history was noncontributory. He did not have any disturbance in his vision. He had taken some over the counter medications (possibly some analgesics and antimicrobials), but had no relief.

On inspection, erythema was evident on the left side of the nose, extending superiorly from the middle of the forehead to the tip of the nose inferiorly, medially from the midline of the face, to laterally up to the level of the pupil of the eye on the left side. Scabbing was evident throughout the region of erythema [Figure 1]. There was abnormal twitching of the left eye, which was accompanied by tearing, conjunctival hyperemia, and palpebral edema [Figure 2]. There was erythema of the nasal turbinates on the left side.
Figure 1: Frontal view of the patient

Click here to view
Figure 2: Left eye of the patient

Click here to view


On palpation, nonpitting edema was felt on the left side of the nose, over the erythematous area, and tremendous pain was elicited. Apparently, some infection of virus (such as herpes zoster) or bacteria (such as impetigo) was suspected. However, the patient did not have any fever. The pain evoked by touching the left side of the nose subsided with nasociliary nerve block, through the medial orbital approach. Based on these clinical presentations, the diagnosis of Charlin's syndrome was made.

Treatment

The patient was prescribed gabapentin (200 mg, three times daily for 3 weeks), amoxicillin (500 mg, thrice daily for 5 days), metronidazole (400 mg, thrice daily for 5 days), acyclovir (200 mg, five times daily for 7 days), and tramadol (50 mg whenever needed). The patient recovered after 5 days and was discharged from the hospital. Consecutive check-up visits revealed no signs of recurrence.


   Discussion Top


Charlin's syndrome is a very uncommon pathological condition, characterized by severe unilateral pain, generated by irritation of the ciliary ganglion,[6] involving the outer aspect of the nose, orbit, and frontal region.[4] The site of pain depends on the distribution of the nasal branch of the trigeminal nerve having neuritis, and accordingly, the pain may be distributed above and outside the nose, above the inner canthus, or along the inferior angle of the medial tarsal ligament.[2] Associated symptoms such as unilateral vasomotor obstruction of the nasal fossae, sneezing, hydrorrhoea, cutaneous hyperaesthesia and conjunctival hyperemia, keratitis, corneal ulcer, and iridocyclitis may be detected.[4] There might be slight inflammatory swelling of upper eyelid, photophobia, ciliary, and conjunctival infection.[2]

Though the exact cause is unknown,[7] sinusitis, high deviations of the nasal septum, infection, and intoxication (alcohol, tobacco, renal, or hepatic dysfunction, diabetes) have been hypothesized to be the contributing factors.[4] Similarly, the patient suffered from pain in his left lateral nostril and left eye associated with slight inflammatory swelling of ipsilateral upper eyelid. There was rhinorrhea and rhinitis on the left side. Photophobia was not evident in the patient. Patient gave an occasional history of sneezing.

The diagnostic criteria for Charlin's syndrome

A. Stabbing pain lasting seconds to hours on one side of the nose, radiating upwards to the medial frontal region and fulfilling criteria B and C.[8] In the presented case, the pain was restricted to patient's left side of the nose radiating to the forehead and the left eye. Each episode of stabbing pain lasted for 10-45 min.

B. Pain precipitated on touching the lateral aspect of the ipsilateral nostril.[8] Similarly, touching the patient's left side of the nose generated lancinating pain.

C. Pain abolished by block or section of the nasociliary nerve, or by the application of cocaine to the nostril on the affected side.[8] In our case, the pain subsided with a nasociliary nerve block.

Differential diagnosis and special investigations

The differential diagnosis includes cluster headache, temporal arteritis, trigeminal neuralgia involving the first division of the trigeminal nerve, demyelinating disease, and chronic paroxysmal hemicrania. The pain of chronic paroxysmal hemicrania lasts much longer than the pain of Charlin's syndrome. Trigeminal neuralgia involving the first division of the trigeminal nerve is uncommon and is characterized by trigger areas and tic-like movements. Demyelinating disease is generally associated with other neurological findings, including optic neuritis and other motor and sensory abnormalities.[7] Trigeminal autonomic cephalgias, a group of syndromes with headaches, associated with combinations of lacrimation, nasal congestions, and eyelid edema,[9] can also be considered under differential diagnosis. However, unlike Charlin's syndrome, the pain is primarily distributed unilaterally in the head.

Special investigations for Charlin's syndrome may include the following: Magnetic resonance imaging may be helpful in ruling out intracranial and brainstem pathological conditions (such as tumors and demyelinating disease and aneurysms). Radionuclide bone scanning and plain radiography can rule out fracture or bony abnormality (such as metastatic disease). Intraocular pressure should be measured if glaucoma is suspected.[7]

Treatment

High dose anticonvulsants, such as carbamazepine and gabapentin, should be the first line of treatment. Nasociliary ganglion block with local anesthetic and steroid may be administered to unresponsive patients. In case of an associated sleep disturbance and depression, a single bedtime dose of tricyclic antidepressants such as nortriptyline (25 mg) may be given.[7]

In our case, the patient responded well to gabapentin. Antimicrobials were administered as microbial cause of neuralgia was suspected due to the presence of scabs. Steroids were avoided for the same reason. Tramadol was prescribed for emergency management of pain. The patient responded well to the treatment and did not require any of the other medications mentioned above.


   Conclusion Top


Charlin's syndrome is a pathological condition, where touching the outer aspect of the nostril causes lancinating pain radiating to the medial frontal region. Even though Charlin's syndrome is a rare disease, we should keep it in mind while diagnosing pain arising from the nose, forehead and the internal angle of the eye.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/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.

 
   References Top

1.
Charlin C. Le Syndrome du Nerf Nasal. Ann Ocul 1931;168:86.  Back to cited text no. 1
    
2.
Roy FH. Ocular Syndromes and Systemic Diseases. 4th Ed. Coralville, Iowa: FEP international; 2008. p. 233.  Back to cited text no. 2
    
3.
Syndromes and eponimes in Otolaryngology. Romanian J Rhinol 2011;1:223.  Back to cited text no. 3
    
4.
Farri A, Enrico A, Farri F. Headaches of otolaryngological interest: Current status while awaiting revision of classification. Practical considerations and expectations. Acta Otorhinolaryngol Ital 2012;32:77-86.  Back to cited text no. 4
[PUBMED]    
5.
Siccoli MM, Bassetti CL, Sándor PS. Facial pain: Clinical differential diagnosis. Lancet Neurol 2006;5:257-67.  Back to cited text no. 5
    
6.
Yee AJ. Medical Eponyms. Available from: http://eponyms.net/eponyms_2009-08-14.pdf. Last accessed on 2016 Jul 25.  Back to cited text no. 6
    
7.
Waldman SD. Atlas of Uncommon Pain Syndromes. 3rd Ed. Philadelphia, PS: Elsevier Saunders; 2013. p. 9-10.  Back to cited text no. 7
    
8.
The International Classification of Headache Disorders, 2nd Edition. Headache Classification Subcommittee of the International Headache Society. Oxford, UK: Blackwell Publishing; 2004. p. 128.  Back to cited text no. 8
    
9.
Goadsby PJ. Trigeminal autonomic cephalgias (TACs). Acta Neurol Belg 2001;101:10-9.  Back to cited text no. 9
[PUBMED]    


    Figures

  [Figure 1], [Figure 2]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

   Abstract Introduction Case Report Discussion Conclusion Article Figures
  In this article
 References

 Article Access Statistics
    Viewed207    
    Printed0    
    Emailed0    
    PDF Downloaded60    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]