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A severe case of cheilitis granulomatosa: clinical-pathologic findings and management

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Oral and Maxillofacial Surgery Aims and scope Submit manuscript

Abstract

Introduction

Cheilitis granulomatosa is an idiopathic granulomatous disorder which is rare and cosmetically disturbing. It presents with a persistent painless labial swelling which obscure etiology. Diagnosis is mainly based on the histopathological features following biopsy. There is no definitive treatment of this disease, reason being its lack of well-defined etiology. It has been proven largely resistant to most of the treatment options.

Case Description

A 28-year-old man presented with the labial swelling which was confirmed to be cheilitis granulomatosa following histopathologic examination. We have successfully managed this case using intralesional steroids.

Conclusion

Intralesional steroids can be used successfully in management of cheilitis granulomatosa.

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References

  1. Miescher G (1945) Uber essentielle granulomatose makrocheilie (cheilitis granulomatosa). Dermatologica 91:57–85

    Article  Google Scholar 

  2. Goel S, Khorate M, Nahar P, Ahmed J (2010) Cheilitis granulomatosa – an uncommon clinicopathological entity: a case report. Journal of cancer science and therapy 2(4):086–088

    Article  Google Scholar 

  3. Camacho-Alonso F, Bermejo-Fenoll A, Lopez-Jornet P (2004) Miescher’s cheilitis granulomatosa. A presentation of five cases. Med Oral Patol Oral Cir Bucal 9:425–429

    Google Scholar 

  4. Wiesenfeld D, Ferguson MM, Mitchell DN, MacDonald DG, Scully C, Cochran K et al (1985) Oro-facial granulomatosis: a clinical and pathological analysis. Q J Med 54:101–113

    CAS  PubMed  Google Scholar 

  5. Grave B, McCullogh M, Wiesenfeld D (2009) Orofacial granulomatosis – a 20 year review. Oral Dis 15(1):46–51

    Article  CAS  PubMed  Google Scholar 

  6. Sciubba JJ, Said-Al-Naief N (2003) Orofacial granulomatosis: presentation, pathology and management of 13 cases. J Oral Pathol Med 32:576–585

    Article  PubMed  Google Scholar 

  7. Seghers A-K, Grosber M, Urbain D, Mana F (Apr-Jun 2019) Cheilitis granulomatosa and Crohn’s disease : a case report. Acta Gastroenterol Belg 82(2):326–328

    PubMed  Google Scholar 

  8. Nazzaro G, Muratori S, Carrera CG, Coggi A, Gianotti R (2015) Cheilitis granulomatosa associated with lupus eritematosus discoid and treated with methotrexate: report of a case. An Bras Dermatol 90(3):200–202

    Article  PubMed  PubMed Central  Google Scholar 

  9. Critchlow WA, Chang D (2014) Cheilitis Granulomatosa: a review. Head and Neck Pathol 8:209–213

  10. Hornstein OP (1973) Melkersson Rosenthal syndrome: a neuro-mucocutaneous disease of complex origin. Curr Probl Dermatol 5:117–156

    Article  CAS  PubMed  Google Scholar 

  11. Kuno Y, Sakakibara S, Mizuno N (1992) Actinic cheilitis granulomatosa. J Dermatol 19(9):556–562

    Article  CAS  PubMed  Google Scholar 

  12. Wehl G, Rauchenzauner M (2018) A systematic review of the literature of the three related disease entities cheilitis granulomatosa, orofacial granulomatosis and Melkersson-Rosenthal syndrome. Curr Pediatr Rev 14(3):196–203

    Article  PubMed  Google Scholar 

  13. Schlarbaum JP, Kimyon RS, Liou YL, Voller LM, Seyfer SJ, Warshaw EM (2019) Hold the spice: allergy to garlic and sulfites—possible relevance in a patient with cheilitis granulomatosa. Contact Dermatitis 81:1–2. https://doi.org/10.1111/cod.13345

    Article  Google Scholar 

  14. Zimmer WM, Rogers RS 3rd, Reeve CM, Sheridan PJ (1992 Nov) Orofacial manifestations of Melkersson-Rosenthal syndrome. A study of 42 patients and review of 220 cases from the literature. Oral Surg Oral Med Oral Pathol 74(5):610–619

    Article  CAS  PubMed  Google Scholar 

  15. Oliver V, Lacour JP, Castanet J, Perrin C, Ortonne JP (2000) Cheilitis granulomatosa in a child. Arch Pediatr 7:274–277

    Article  Google Scholar 

  16. Park HS, Park ES, Park KC, Cho KH, Youn SW (2008) Chronic idiopathic macrocheilia associated with simple lip enlargement and salivary gland hyperplasia. J Dermatol 35:234–237

    Article  PubMed  Google Scholar 

  17. Allen CM, Camisa C, Hamzeh S (1990) Cheilities granulomatosa: report of six cases and review of literature. J Am Acad Dermatol 23:444–450

    Article  CAS  PubMed  Google Scholar 

  18. Ratzinger G, Sepp N, Vogetseder W, Tilg H (2007) Cheilitis granulomatosa and Melkersson-Rosenthal syndrome: evaluation of gastrointestinal involvement and therapeutic regimens in a series of 14 patients. J Eur Acad Dermatol Venereol 21:1065–1070

    Article  CAS  PubMed  Google Scholar 

  19. Kano Y, Shiohara T, Yagita A, Nagashima M (1992) Treatment of recalcitrant cheilitis granulomatosa with metronidazole. J Am Acad Dermatol 27:629–630

    Article  CAS  PubMed  Google Scholar 

  20. Ishiguro E, Hatamochi A, Hamasaki Y, Ishikawa S, Yamazaki S (2008) Successful treatment of granulomatous cheilitis with roxithromycin. J Dermatol 35:598–600

    Article  PubMed  Google Scholar 

  21. Sutharaphan T, Chanprapaph K, Vachiramon V (2019 Sep 27) Unsuccessful treatment of cheilitis granulomatosa with oral methotrexate. Case Rep Dermatol 11(3):249–255. https://doi.org/10.1159/000503138 eCollection Sep-Dec 2019

  22. Barry O, Barry J, Langan S, Murphy M, Fitzgibbon J, Lyons JF (2005) Treatment of granulomatous cheilitis with infliximab. Arch Dermatol 141:1080–1082

    Article  PubMed  Google Scholar 

  23. Jones E, Callen JP (1990) Hydroxychloroquine is effective therapy for control of cutaneous sarcoidal granulomas. J Am Acad Dermatol 23:487–489

    Article  CAS  PubMed  Google Scholar 

  24. Belfield KD, Tichy EM (2018) Review and drug therapy implications of glucose-6-phosphate dehydrogenase deficiency. Am J Health Syst Pharm 75(3):97–104

    Article  CAS  PubMed  Google Scholar 

  25. Seth S, Khan DA. The comparative safety of multiple alterna- tive agents in refractory chronic urticaria patients. J Allergy Clin Immunol Pract 2017; 5(1): 165–170.e2

  26. Gronostay A, Hellmich L , Rasokat H , Hunzelmann N. Coincidence of cheilitis granulomatosa and chronic spontaneous angioedema: successful treatment with dapsone and omalizumab. Dtsch Dermatol Ges. 2020 May 24. doi: https://doi.org/10.1111/ddg.14104, 18, 611, 613

  27. Gu L , Huang DY , Fu CJ , Wang ZL , Liu Y , Zhu GX. Successful treatment of cheilitis granulomatosa by intralesional injections of pingyangmycin plus dexamethasone. Eur J Dermatol. 2016 Dec 1;26(6):627–628. doi: https://doi.org/10.1684/ejd.2016.2863

  28. Bacci C, Valenta ML (2010) Successful treatment of cheilitis granulomatosa with intralesional injection of triamcinolone. J Eur Acad Dermatol Venereol 24:363–364

    Article  CAS  PubMed  Google Scholar 

  29. Cerimele D, Serri F (1963) Intralesional injection of triamcinolone in the treatment of cheilitis granulomatosa. Arch Dermatol 72:695–696

    Google Scholar 

  30. Eisenbud L, Hymowitz SS, Shapiro R (1971) Cheilitis granulomatosa: report of case treated with injection of triamcinolone acetonide aqueos suspension. Oral Surg Oral Med Oral Pathol 32:384–389

    Article  CAS  PubMed  Google Scholar 

  31. Kawakami T, Fukai K, Sowa J, Ishii M, TeramaeH, Kanazawa K. Case of cheilitis granulomatosa associated with apical periodontitis. J Dermatol. 2008; 35: 115-119

  32. Worsaae N, Cristensen KC, Schiodt M, Reibel J (1982) Melkersson-Rosenthal syndrome and cheilitis granulomatosa. A clinicopathologic study of thirty three patients with special reference to their oral lesions. Oral Pathol 54:404–413

    Article  CAS  Google Scholar 

  33. Oliver DW, Scott MJ (2002) Lip reduction cheiloplasty for Miescher’s granoulomatous macrocheilitis (Cheilitis granulomatosa) in childhood. Clin Exp Dermatol 27:129–131

    Article  PubMed  Google Scholar 

  34. Innocenti A, Innocenti M, Taverna C, Melita D, Mori F, Ciancio F, de Giorgi V, Parronchi P, Vultaggio A, Matucci A (2017) Miescher’s cheilitis: surgical management and long term outcome of an extremely severe case. Int J Surg Case Rep 31:241–244. https://doi.org/10.1016/j.ijscr.2017.01.062 Epub 2017 Jan 30

    Article  PubMed  PubMed Central  Google Scholar 

  35. Krutchoff D, Randal J (1978) Cheilitis granulomatosa: successful treatment with combined local triamcinolone injections and surgery. Arch Dermatol 114:1203–1206

    Article  Google Scholar 

  36. Critchlow WA, Chang D (2014) Cheilitis Granulomatosa: a review. Head and Neck Pathol 8:209–213

    Article  Google Scholar 

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Authors and Affiliations

  1. Department of Oral and Maxillofacial Surgery, Awadh Dental College, Jamshedpur, Jharkhand, India

    Abhilasha Yadav

  2. Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, Maharashtra, India

    Abhilasha Yadav, Anuj Jain & Rajiv Borle

  3. Consultant Oral and Maxillofacial Surgeon, Nagpur, Maharashtra, India

    Anuj Jain

  4. Department of General Surgery, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, Maharashtra, India

    Suhas Jajoo

Authors
  1. Abhilasha Yadav
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  2. Anuj Jain
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  3. Rajiv Borle
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  4. Suhas Jajoo
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Correspondence to Anuj Jain.

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Yadav, A., Jain, A., Borle, R. et al. A severe case of cheilitis granulomatosa: clinical-pathologic findings and management. Oral Maxillofac Surg 24, 521–526 (2020). https://doi.org/10.1007/s10006-020-00875-2

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  • DOI: https://doi.org/10.1007/s10006-020-00875-2

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