Successful treatment of Miescherís cheilitis in Melkersson-Rosenthal syndrome with betamethasone injections and doxycycline
Lamia Oudrhiri, Soumiya Chiheb, Farida Marnissi, Soumaya Zamiati, Hakima Benchikhi
The Pan African Medical Journal. 2012;13:75. doi:10.11604/pamj.2012.13.75.1895

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Case report

Successful treatment of Miescherís cheilitis in Melkersson-Rosenthal syndrome with betamethasone injections and doxycycline

Cite this: The Pan African Medical Journal. 2012;13:75. doi:10.11604/pamj.2012.13.75.1895

Received: 20/07/2012 - Accepted: 30/08/2012 - Published: 09/12/2012

Key words: Miescherís cheilitis granulomatosa, Melkersson-Rosenthal syndrome, treatment

© Lamia Oudrhiri et al. The Pan African Medical Journal - ISSN 1937-8688. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Available online at: http://www.panafrican-med-journal.com/content/article/13/75/full

Corresponding author: Lamia Oudrhiri, Department of Dermatology Venerology Ibn Rusd UHC, Casablanca, Morocco (lamiaoudrhiri@live.fr)


Successful treatment of Miescherís cheilitis in Melkersson-Rosenthal syndrome with betamethasone injections and doxycycline

 

Lamia Oudrhiri1, Soumiya Chiheb1, Farida Marnissi2, Soumaya Zamiati2, Hakima Benchikhi1

 

1Department of Dermatology Venerology Ibn Rusd UHC, Casablanca, Morocco, 2Department of Pathology Ibn Rushd UHC, Casablanca, Morocco

 

 

&Corresponding author
Lamia Oudrhiri, Department of Dermatology Venerology Ibn Rusd UHC, Casablanca, Morocco

 

 

Introduction

Treatment of Miescherís cheilitis granulomatosa integrated or not in Melkersson-Rosenthal syndrome is a real therapeutic challenge. We report a case of 19- year-old girl successfully treated by association of local betamethasone injections and doxycycline.

 

 

Patient and observation

A 19- year-old girl presented a 5-year history of a painless enlargement of upper lip. Swelling was initially asymmetrical and intermittent then became permanent and diffuse. The patient received dapsone and oral steroids without any improvement. Clinical examination found a diffuse swelling of upper lip (Figure 1) associated to lingua plicata and facial asymmetry. Neurological examination objectified facial nerve palsy. Diagnosis of Melkersson-Rosenthal syndrome was retaind because of association of cheilitis, lingua plicata and facial paralysis. Labial mucosa biopsy showed non-necrotizing giganto-epithelioid granuloma confirming Miescher's cheilitis granulomatosa (Figure 2). A specialized management of caries and dental foci of infection was performed before starting treatment.

We prescribed intra-lesional betamethasone injections (7 mg / ml): 1 ml per injection (Figure 3) once a month for three months associated to a single daily dose of doxycycline 200 mg for 3 months. An anesthetic cream with lidocaine and prilocaine (EMLA 5%) was applied one hour before each injection to reduce pain. From the first month of treatment, an important improvement of cheilitis was seen. Gradual and permanent reduction of the upper lip volume was observed after three months of treatment (Figure 4). One year follow up objectified no reactivation of cheilitis.

 

 

Discussion

We report a case of Miescherís cheilitis in Melkersson-Rosenthal syndrome treated successfully by the association betamethasone injections and doxycycline. The use of betamethasone was decided because of non availability of triamcinolone. Intralesional injections of corticosteroids are very painful. A regional anesthesia of a nerve block before administration of triamcinolone may be recommended [1]. In our case local anesthetic cream allowed a good tolerance of treatment.

The management of patients with cheilitis granulomatosa remains a challenge and should be related to the severity of the symptoms .Various therapeutic strategies have been proposed in literature including clofazimine, systemic or intralesional steroids alone or associated to dapsone and in some isolated cases; hydroxychloroquine, metronidazole, thalidomide and infliximab [2-4]. However, treatment is empirical.

The use of cycline is based on its in vitro ability to inhibit granuloma formation by inhibition of protein kinase C. This result provided the successful use of minocycline in the treatment of granulomatous dermatitis [5-7]. The association minocycline and corticosteroids would be more efficient in reducing cheilitis recurrences [8]. Similar combination has been successfully used in children and adult patients [8,9]. Camacho et al pointed out the effectiveness of a single injection of triamcinolone immediately after reduction cheiloplasty associated with gradually decreasing doses of tetracycline over a period of 6 months in 27 adult cases [10].

 

 

Conclusion

The association betamethasone and doxycycline is a very interesting alternative therapy for unaesthetic, displaying and resistant cheilitis.

 

 

Competing interests

The authors declare no competing interests.

 

 

Authorsí contributions

All the authors have contributed to this manuscript in ways that comply to the ICMJE authorship criteria. All the authors have read and approved the final version of the manuscript.

 

 

Figures

Figure 1: Diffuse swelling of the upper lip.

Figure 2: Labial mucosa biopsies showed non-necrotizing giganto-epithelioid granuloma

Figure 3: Intralesional betamethasone injection

Figure 4: Result after three months of treatment

 

 

References

  1. Sakuntabhai A, MacLeod R, Lawrence C. Intralesional steroid injection after nerve block anesthesia in the treatment of orofacial granulomatosis. Arch Dermatol. 1993; 129 (4): 477-480. This article on PubMed

  2. Rose AE, Leger M, Chu J, Meehan S. Cheilitis granulomatosa. Dermatol Online J. 2011 ;17 (10):15. This article on PubMed

  3. Bacci C, Valente ML. Successful treatment of cheilitis granulomatosa with intralesional injection of triamcinolone. J Eur Acad Dermatol Venereol. 2010 Mar;24(3):363-4. This article on PubMed

  4. Sobjanek M, Wlodarkiewicz A, Zelazny I et al. Successful treatment of MelkerssonĖRosenthal syndrome with dapsone and triamcinolone injections. J Eur Acad Dermatol Venerol. 2008; 22(8): 1028-1029

  5. Webster GF, Toso SM, Hegemann L. Inhibition of a model of in vitro granuloma formation by tetracyclines and ciprofloxacin: involvement of protein kinase C. Arch Dermatol. 1994; 130 (6):748-752. This article on PubMed

  6. Sapadin AN, Fleischmajer R. Tetracyclines: nonantibiotic properties and their clinical implications. J Am Acad Dermatol. 2006; 54 (2): 258-265. This article on PubMed

  7. Tsankov N, Broshtilova V, Kazandjieva J. Tetracyclines in dermatology. Clin Dermatol. 2003 Jan-Feb;21(1):33-9. This article on PubMed

  8. Stein SL, Mancini A J. MelkerssonĖRosenthal syndrome in childhood: successful management with combination steroid and minocycline proved effective and well tolerated in two children. J Am Acad Dermatol. 1999; 41 (5): 746-748. This article on PubMed

  9. Lynde CB, Bruce AJ, Orvidas LJ, Rogers R S, Depry JL. Cheilitis granulomatosa treated with intralesional corticosteroids and anti-inflammatory agents. J Am Acad Dermatol. 2011 Sep;65(3):e101-2. This article on PubMed

  10. Camacho F, GarcŪa-Bravo B, Carrizosa A. Treatment of Miescherís cheilitis granulomatosa in MelkerssonĖRosenthal syndrome. J Eur Acad Dermatol Venereol. 2001 Nov;15(6):546-9. This article on PubMed

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 


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