Cheilitis Query

September 2017
The patient is a 70 yo Caucasian who has lived on Moorea, French Polynesia, for the past 50 years.  She contacted us recently about her painful lips because there is no dermatologist available to her at present.  Here is her anamnesis:

In early July when I went to Montreal, my lips started to bother me. I thought maybe it was 18 hours on a plane, or even maybe it was a sunburn from being in the pool with my grandchildren in sunny Vancouver a couple weeks before. It didn't subside and I bought several lip therapies - cocoa butter, Vaseline, Aquaphor. When I came home I used a mild steroid ointment for a couple weeks, but to no effect. I now carry Aquaphor with me all the time and apply it constantly. Chapstick with SPF (from a friend in the States) stings my lips, as does toothpaste. My lips are not chapped, as in flaky or peeling, but they feel and look burnt, even blistery sometimes, and they can feel severely tight, dry and very sore. Actually, my upper lip is not as involved as my lower lip, and the corners are not affected. 

Photo sent by patient to VGRD

Diagnosis: This appears to be actinic cheilitis or possibly allergic/irritant cheilitis.  Strangely,  the patient got more sun in Vancouver than she does in French Polynesia!

What are your thoughts?

Update (April 2018):

(from the patient) I want to share with you the results of a recent experiment I conducted unwittingly. Our daughter was visiting Tahiti for the last week, and we went to the beach several days in a row. Although I wore a hat and Vanicream lip sunscreen, I got too much sun and my lower lip has been on fire. I think actinic cheilitis was an early guess last year, and I have NO DOUBT that it was correct. I'm using Vaseline, of course, and the betamethasone dipropionate ointment after nothing else worked. I'm happy not to be puzzled, confused and freaked out this time around.  Note:  I think we may be dealing with a case of actinic prurigo of the lips (see references 3 and 4 below)

References:

1. Actinic cheilitis: a treatment review.

Shah AY, Doherty SD, Rosen T.

Abstract:  All other factors being equal, the presence of actinic cheilitis, a pre-invasive malignant lesion of the lips, doubles the risk of squamous cell carcinoma developing in this anatomic area. Various forms of local ablation, immunomodulation and surgical extirpation have been proposed as therapeutic interventions. This paper critically evaluates the available medical literature to highlight the evidence-based strength of each recommended therapy for actinic cheilitis. Vermilionectomy remains the gold standard for efficacy; trichloroacetic acid application is easy and convenient, but the least efficacious overall.

2. Contact allergy in cheilitis.

O'Gorman SM, Torgerson RR. Int J Dermatol. 2016 Jul;55(7):e386-91.

BACKGROUND: Recalcitrant non-actinic cheilitis may indicate contact allergy.

CONCLUSIONS: Contact allergy is an important consideration in recalcitrant cheilitis. Fragrances, antioxidants, and preservatives dominated the list of relevant allergens in our patients. Nickel and gold were among the top 10 allergens. Almost half (45%) of these patients had a final diagnosis of ACC. Patch testing beyond the oral complete series should be undertaken in any investigation of non-actinic cheilitis.

3. Actinic Prurigo Cheilitis: A Clinicopathologic Review of 75 Cases.

Plaza JA, et al. Am J Dermatopathol. 2016 Jun;38(6):418-22.

4. Actinic prurigo of the lip: Two case reports.

Miranda AM. World J Clin Cases. 2014 Aug 16;2(8):385-90. Free Full Text.

Perplexing Recurrent Cheilitis

Presented by Henry Foong
Ipoh, Malaysia

 

Abstract: A 20-year-old man presented with recurrent cheilitis 

HPI: A 20-year-old student presented with recurrent peeling of the lips for about 3 years. It usually began on the upper lip then involved the lower lip. Then the cycle repeated. The entire process takes about 3 weeks. He has no known drug allergy. He denied any lip smacking. He was using a moisturising non-SLS tooth paste.

Examination showed peeling of the lower and upper lip as a single friable sheet. His oral cavity and genitalia were unremarkable.

Clinical Images: 

Patch Tests:                                 

Gold sodium thiosulphate  ++ at 48 and 96 hours;

Iodopropynyl butylcarbamate  + at 48 and – at 96 hours

Sodium bisulphide + at 48 and 96 hours

Thimerosal +/- at 48 and 96 hours

Histopathology: The sections show a fragment of tissue surfaced by parakeratinised stratified squamous epithelium. The underlying connective tissue is moderately collagenous with mild chronic inflammatory cells infiltration and a few small blood vessels. A few lobules of minor salivary glands are observed. No granuloma seen in the sections examined. Final Path Diagnosis: Lower labial mucosa: Histologically non-specific 

Diagnosis: Chronic cheilitis

Reason for presentation  

Despite avoiding all the trigger factors such as lip smacking and use of non-SLS toothpaste, his symptoms persisted. The patch test results are probably irrelevant in his case. In a study from Singapore, toothpastes were the commonest cause of allergic contact cheilitis in males. Ricinoleic acid and the patient's own lip preparations were the commonest relevant contact allergens. The absence of granuloma practically make granulomatous cheilitis unlikely.

Questions   

What is the most likely diagnosis? Could this be a case of exfoliative cheilitis? Exfoliative cheilitis, a rare, localized condition, is a chronic superficial inflammatory condition that is characterized by regular peeling of a superficial excessive layer of keratin. the cause of this condition is unknown but may be associated with depressive illness. Two other differential diagnosis comes to mind - pemphigus vulgaris and Crohn's disease.

References

1. Mani SA, Shareef BT. Exfoliative cheilitis: report of a case. J Can Dent Assoc. 2007 Sep;73(7):629-32. Abstract.  Full Free Text.

2. Lim SW, Goh CL. Epidemiology of eczematous cheilitis at a tertiary dermatological referral centre in Singapore.Contact Dermatitis. 2000 Dec;43(6):322-6. Abstract.

3.  Contact allergy in cheilitis.  O'Gorman SM, Torgerson RR.  Int J Dermatol. 2015 Nov 6.  Abstract. (See Dr. Sharquie's comments)

4. Exfoliative Cheilitis DermNet-NZ. (excellent synopsis)

Keywords   

chronic cheilitis, exfoliative cheilitis

Severe Recurrent Cheilitis

Presented by Nyoman Sukano

Singaraja, Bali Indonesia

A 20 year-old student complains of recurrent peeling of the lips for more than a year. It usually begins on the upper lip then involves the lower lip. Then the cycle repeats. The entire process takes about 3 weeks. He is known to have Hemophilia A and on regular follow up for factor VIII.  He has no known drug allergy. Denies any wetting of lips or lip smacking. Uses a moisturizing tooth paste.

Examination was unremarkable except for peeling of the lower and upper lip as a single friable sheet. The  oral cavity was unremarkable.  His genitalia and rest of cutaneous exam was normal.

Clinical Images:

Lab:  Patch test was done but was negative (results may not be very reliable as he was on oral prednisolone 10mg daily during the test; but did not let us know).  No other test done.

Treatment: We have tried Vaseline and  mometasone ointment but they did not work.

Diagnosis: Is this cheilitis?

Thanks for your thoughts.

Cheilitis Oscura

Abstract:  59 yo woman with eight month history of cheilitis.

HPI: The patient is a 59 yo woman with an eight month history of a pruritic dermatitis which began on the upper lip. It spread to the lower lip and recently to some fingers. For dental hygiene, she has used Crest toothpaste, a white ceramic nasal irrigator, dental floss, a WaterPic and a Sonic Dental Care apparatus on a daily basis.  She has changed this protocol somewhat over the past few months.  Initially, she used Desonide ointment for three to four weeks with minimal help.  She also used clobetasol ointment 0.05% for 10 days with complete clearing. After a protocol of erythromycin gel, steroid ointment, protopic 0.03% the iritation largely disappeared. However, she continues to get flare ups with no apparent cause. Symptoms always include burning, itching and redness. She uses Vaseline on a regular basis to moisten and sooth her lips. A similar dermatitis has appeared around the nail of her right ring finger, and has lingered there for 3 months. In addition, her left ear has several similar spots, and other similar spots have come and gone around her eye lids, eye brow and forehead. Most recently, she has used tacrolimus 0.03% ointment with minimal help, and for the last few weeks only Vaseline, which seems to help the most.  Because of the concern for "steroid addiction" of the facial skin, she only used topical steroids for short periods and only under close supervision.  The process waxes and wanes unpredictably.

O/E:  The process is localized, erythematous and scaly.  The secondary lesions on the fingers and ears look similar.

Clinical Photos:
12/11/11

4/8/12

4/28/12

Laboratory:

Patch Test (T.R.U.E.) all 29 negative.  More focused patch testing is indicated.

Lip culture:  Staph epidermitis and alpha hemolytic strep (interpreted as normal skin organisms)

Biopsy:  Considered, but not done at this time.

Diagnosis and Discussion: Initially, I presumed this to be an allergic contact dermatitis.  I suspect that part of her regimen is the culprit. The sharp border is unusual and could suggest a koebnerized psoriasiform process from an oral hygiene devices that comes in contact with the area.  In children, lip licking would be considered (lick eczema) but the history here does not support that diagnosis.  If the process recurs, I would ask patient to let it develop and then biopsy the lesion.  If that was not helpful, then patch testing with a dental tray would settle the issue of allergic contact dermatitis. 

Questions: Have you encountered similar cases? What are your thoughts?

Reference:  
1.  This is a helpful reference which goes into the many causes of contact dermatitis around the mouth and lips.  Available Free Full Text.  Andrew Scheman, MD, et. al.  Part 3 of a 4-part series Lip and Common Dental Care Products: Trends and Alternatives.  Data from the American Contact Alternatives Group.  Clin Aesthet Dermatol. 2011 September; 4(9): 50–53. 

2.  Here is another useful reference on persistent cheilitis, also available Free Full Text. 

Cheilitis in a Young Woman

Abstract: 1.5 year history of cheilitis in a 26 yo woman
HPI: This is a a 23 yo esthetician who first developed cheilitis on her honeymoon in Mexico. She was using many lip balms at the time. These were discontinued and she found toothpaste without cinnamates. The process continued to flare. While initially on upper and lower lips, it is now just on the lower lip. She has read a lot on the subject and has many concerns. The patient has had a problem with anxiety since her father's death when she was 14 and sees a therapist. Cool compresses and fluocinalone 0.025% ointment control the problem fairly well; but it flares when she stops this. At one point, a KOH prep was positive for Candida (but that was when she'd been using a optical corticosteroid and this resolved quickly with ketoconazole cream).
O/E: Recently, the process is located on the lower lip. Here there is erythema, some induaation and scaling.
Clinical Photos: 1/10/1010










This photo was taken on Feb. 8, 2010 after a month off usual lip products and use of Vaseline and/or fluocinalone ointment.

Pathology: A 3 mm punch biopsy was taken from the lower lip.
This shows "confluent scale crust containing neutrophils, acanthosis with spongiosis and a dense lichenoid infiltrate. No granulomatous changes. The pathologist felt that these changes were non-diagnostic but "consistent with cheilitis glandularis."
Diagnosis: Cheilitis. In the ddx is contact, cheilitis glandularis and factitial cheilitis.
Discussion: I am not comfortable with a disgnosis of cheilitis glandularis here. Contact and factitial etiologies could still play a role. Patch testing will be done, but if negative and the problem persists consideration to having more in-depth patch testing may be given. We are also working with patient to stop licking and chewing lips.
Questions: What are your thoughts re: 00etiology here?
Addendum: A few months after this case was presented, the patient recalled that she had received a new dental retainer shortly befor the cheilitis began. Her old retainer was plastic and the new one was metal. She stopped using this new retainer and her cheilitis disappeared. Thus, a metal sensitivity (most likely nickel) was the culprit.
References:
1. Nico MM, Nakano de Melo J, Lourenço SV. Cheilitis glandularis: A clinicopathological study in 22 patients. J Am Acad Dermatol.. [Epub ahead of print]
Department of Dermatology, Medical School, São Paulo, Brazil.
BACKGROUND: Cheilitis glandularis (CG) is a condition in which thick saliva is secreted by minor labial salivary glands and adheres to a swollen lip causing discomfort to the patient. Most publications refer to single case reports or small case series. OBJECTIVE: We sought to report and to analyze clinical, pathological, and therapeutic data on 22 patients with CG seen at the department of dermatology at our university. METHOD: Retrospective data about 22 patients with CG are reviewed and presented. RESULTS: Seventeen patients were male and 5 were female. All were fair skinned, including 6 albino individuals. Several of them presented significant signs of photodamage on the lips. Surgical treatment was performed in 10 severely affected patients and consisted of a vermilionectomy followed by minor salivary gland removal. Histopathological study revealed various degrees of chronic sialadenitis and vermilion epithelial changes. Superficially invasive and in situ squamous cell carcinoma of the vermilion was detected in 3 cases. LIMITATIONS: Biopsy and surgery were not performed in all patients. CONCLUSIONS: CG is strongly related to sun sensitivity and may be more severe in albino patients. The swollen, sun-exposed lip may become more susceptible to the occurrence of squamous cell carcinoma.

2. Aydin E, Gokoglu O, Ozcurumez G, Aydin H. Factitious cheilitis: a case report. J Med Case Reports. 2008 Jan 29;2:29.
Baskent University Department of Otolaryngology, Ankara, Turkey. erdinca@baskent-ank.edu.tr
INTRODUCTION: Factitious cheilitis is a chronic condition characterized by crusting and ulceration that is probably secondary to chewing and sucking of the lips. Atopy, actinic damage, exfoliative cheilitis, cheilitis granulomatosa or glandularis, contact dermatitis, photosensitivity reactions and neoplasia should be considered in the differential diagnosis of crusted and ulcerated lesions of the lip. CASE PRESENTATION: We present a 56 year-old female with an ulcerated and crusted lesion on her lower lip. The biopsy showed granulation tissue and associated inflammation but no malignancy. Based on the tissue examination and through clinical evaluation the diagnosis of factitious cheilitis was rendered. CONCLUSION: Thorough clinical history, utilization of basic laboratory tests and histopathologic evaluation are required to exclude other diseases and a thoruough psychiatric evaluation and treatment is vital for successful management of these patients.

3. Torgerson RR, Davis MD, Bruce AJ, Farmer SA, Rogers RS 3rd. Contact allergy in oral disease. J Am Acad Dermatol. 2007 Aug;57(2):315-21. Epub 2007 May 25.
Department of Dermatology, Mayo Clinic, Rochester, MN 55905, USA.
BACKGROUND: The role of contact allergy in oral cavity disease processes is unknown. OBJECTIVE: We sought to determine the prevalence of contact allergy to flavorings, preservatives, dental acrylates, medications, and metals in patients with oral disease. METHODS: Patients were tested with an 85-item oral antigen screening series. Data were analyzed retrospectively. RESULTS: We evaluated 331 patients with burning mouth syndrome, lichenoid tissue reaction, cheilitis, stomatitis, gingivitis, orofacial granulomatosis, perioral dermatitis, and recurrent aphthous stomatitis. Positive patch test results were identified in 148 of the 331 patients; 90 patients had two or more positive reactions. Allergens with the highest positive reaction rates were potassium dicyanoaurate, nickel sulfate, and gold sodium thiosulfate. Of the 341 positive patch test reactions, 221 were clinically relevant. LIMITATIONS: No follow-up data were available in this retrospective analysis. CONCLUSION: The positive and relevant allergic reactions to metals, fragrances, and preservatives indicated that contact allergy may affect oral disease.
.

Recurrent Cheilitis in a 37 yo Woman

Abstract: 37 yo woman with 18 year recurrent cheilitis

HPI: This 37 yo woman has had recurrent cheilitis for 18 – 20 years. She works as a medical assistant. At one time, she was thought to be latex sensitive because blowing up balloons makes her feel sick, but a RAST test was negative. The episodes last five to six days. The day before the present episode she had eaten a “Mediterranean Dip” which included cucumber, garlic, feta, tomato, and horseradish. She noted nothing till the next morning when there was mild erythema of upper lip. She may have a history of mild atopic dermatitis. She can recall no meds she took before this or other episodes.

O/E: Erythema and mild crusting of lips and adjacent glabrous skin. Remainder of exam normal. Occasionally she’ll have mild erythema around outer canthi.

Photos:




Lab/Path: N/A

Diagnosis: ? Allergic Cheilitis. Role of Foods? Doubt Fixed Drug Eruption. To me this looks like an allergic contact cheilitis.

Questions: What would be the best way to work this up? This woman has 3 - 4 episodes per year, so it is unlikely that her cheilitis is related to something whe uses daily, and she does not recall anything she applies only intermittently.

Reference:
Allergic contact cheilitis in the United Kingdom: a retrospective study.
Strauss RM, Orton DI.
Am J Contact Dermat. 2003 Jun;14(2):75-7.

Abstract: Environmental and Contact Dermatitis Unit, Amersham Hospital, Whielden Street,
Amersham, Buckinghamshire, HP7 0JD, United Kingdom. strauss@strauss.karoo.co.uk

BACKGROUND: To date, only a few cohorts of patients with allergic cheilitis have
been described, most of them from Australia and Asia. OBJECTIVE: To establish the
prevalence of cheilitis in a UK specialist contact dermatitis clinic and to
identify the most common allergens. METHOD: We analyzed our patch-test database
in a tertiary referral center in the United Kingdom, retrospectively. All
patients presenting with cheilitis over a 19-year period (1982 to 2001) were
included. RESULTS: Data were available from a total of 146 patients. A positive
allergic patch-test reaction was thought to be relevant in 15% of the patients (n
= 22) and to be of possible relevance in 6.8% (n = 10). Of the 22 patients with
relevant allergic results, 95% (n = 21) were women. The most common allergens
included fragrance mix (mainly cinnamaldehyde, oak moss, and isoeugenol) in 41%
of patients, shellac in 18%, colophony in 18%, and Myroxylon pereirae in 14%. For
half of the patients, the allergen was believed to stem from lipsticks or lip
products. Eighteen percent of patients with allergic cheilitis reacted to only
their own products. CONCLUSIONS: Patients should be tested to extended
lipstick/cosmetic vehicle series in addition to standard series. As a significant
percentage of patients react to their own products only, a thorough clinical
history and testing to patients' own products are important.