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

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A severe case of cheilitis granulomatosa: clinical-pathologic findings and management Abhilasha Yadav 1,2 & Anuj Jain 2,3

&

Rajiv Borle 2 & Suhas Jajoo 4

Received: 12 April 2020 / Accepted: 29 June 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020

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. Keywords Cheilitis granulomatosa . Labial swelling . Intralesional steroids . Triamcinolone acetonide

Introduction Cheilitis granulomatosa (CG) is a cosmetically disturbing, rare, unique, and idiopathic granulomatous disorder, initially described by German dermatologist Miescher in 1945 [1] as a distinct clinicopathological entity, characterized clinically by diffuse, non-tender, soft to firm swelling and ulceration of one or both lips [2]. CG has traditionally been considered as a forme fruste of Melkersson–Rosenthal syndrome (MRS), described as the association of recurrent labial edema and/or facial edema, relapsing facial paralysis and fissured tongue. The appearance of the classical triad of MRS is infrequent, however, appearing in approximately 25 to 40% of cases. Generally, patients

* Anuj Jain [email protected] 1

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

2

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

3

Consultant Oral and Maxillofacial Surgeon, Nagpur, Maharashtra, India

4

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

present oligosymptomatic or monosymptomatic forms of the illness, cheilitis granulomatosa being the commonest monosymptomatic form [3]. Orofacial granulomatosis, which was introduced by Wiesenfeld in 1985 [4, 5], is a unifying terminology showing similar clinical features and a broad spectrum of nonnecrotizing granulomatous inflammation in the oral and facial region [6]. The diagnostic dilemma may be further complicated by the fact that CG has often been associated with other orofacial granulomatous disorders like sarcoidosis, Crohn’s disease, atypical tuberculosis, Anderson-Fabry disease, lupus erythematosus discoid, and allergic reactions [2, 7, 8]. Options fo