Actinic cheilitis granulomatosa.

A granulomatous lesion of the lips caused by UVB-photosensitivity is described. A 31-year-old Japanese man developed depigmented, swollen, erythematous lips with some erosions and bloody crusts which were present for 11 months. Histologic examination revealed a dense dermal infiltration composed mainly of lymphoid-histiocytic cells. Suprabasal clefts were associated with the invasion of lymphoid cells around them. Results of patch and photopatch testing were negative. The minimal erythema dose 24 h after irradiation was lower than normal, ranging from 295 to 330 nm. The lesion was reproduced by repeated irradiation with monochromatic light of 310 and 320 nm, but not by exposure to 250, 260, 270, 280, 290, 300, or 330 nm. Topical injection of glucocorticoids and application of sunscreen led to improvement. We propose the name "actinic cheilitis granulomatosa" for this case.     

Granulomatous cheilitis.

Four cases of granulomatous cheilitis, an uncommon disease and one that is difficult to treat, are described. The cause of chronic swelling of the lips has been in dispute for over 50 years. In our material we have excluded sarcoidosis and silica, two of the postulated causes, and we suggest that hypersensitivity is concerned.     

Successful treatment of cheilitis granulomatosa with tranilast

A new anti-allergic drug, tranilast [N-(3′,4′-dimethoxycinnamoyl) anthranilic acid] was used on a 46-year-old Japanese female patient with cheilitis granulomatosa. A swelling of upper lip with repeated recurrence every two weeks was dramatically suppressed by treatment for 3 months. After drug withdrawal, she was free of symptoms for 12 months. The mode of action of tranilast in cheilitis granulomatosa is not understood at the present time.     

Granulomatous cheilitis and Crohn''s disease

A case of granulomatous cheilitis is reported in whom investigations showed intestinal involvement compatible with Crohn's disease, although the patient had no gastrointestinal symptoms. Granulomatous cheilitis (GC) is an uncommon disorder that is characterized by persistent swelling of the lips and a granulomatous histology. Since the report of a case of GC that occurred several years before intestinal lesions appeared,¹ GC has been recognized to be an early manifestation of Crohn's disease. However, to our knowledge only four cases have been reported that initially presented with GC and then developed Crohn's disease.^1–4 Whether GC represents an early manifestation of Crohn's disease or a distinct clinico-pathological entity remains controversial.⁵ We describe a patient with GC who had intestinal lesions compatible with the diagnosis of Crohn's disease, although she had no gastrointestinal symptoms.     

Case of cheilitis granulomatosa associated with apical periodontitis

The etiology of cheilitis granulomatosa is unknown. In some cases, rapid improvement and/or complete elimination of swelling of the lips after dental treatment has been reported. Here, we describe another case of improvement following dental treatment. A 57-year-old woman had developed asymptomatic swelling of the lower lip 2 months previously. Histological examination revealed non-caseous giant cell granulomas. Neither facial nerve palsy nor fissuring of the tongue was present. Patch testing for metal allergy revealed only mild irritation to zinc ion. Although topical corticosteroid ointment and oral tranilast for 4 months were ineffective, rapid and remarkable improvement of the swelling was noted soon after treatment of two lesions of apical periodontitis. Thorough examination for foci of infection is necessary when treating a patient with cheilitis granulomatosa.     

Granulomatous cheilitis and a positive Kveim test

A female patient with a protracted history of multitudinary symptoms more recently developed granulomatous cheilitis (GC) and underwent extensive investigations. Her Kveim test was positive.     

Treatment of Miescher's cheilitis granulomatosa in Melkersson–Rosenthal syndrome

Cheilitis granulomatosa is the most frequent dermatological sign in the Melkersson-Rosenthal syndrome. We reviewed 27 cases of this syndrome diagnosed and treated in the last 20 years. All the patients were surgically treated and received intralesional corticosteroids during surgery and tetracycline hydrochloride (500 mg, twice daily) after the operation to prevent recurrence.     

Surgical treatment of persistent macrocheilia in patients with Melkersson-Rosenthal syndrome and cheilitis granulomatosa

BACKGROUND: Various conservative methods for treatment of labial swelling in patients with cheilitis granulomatosa have been attempted, often with only moderate success and sometimes with persistent disfiguring lip swelling. Severe macrocheilia can produce an unaesthetic facial deformity associated with functional disturbances. In patients with persistent macrocheilia, reduction cheiloplasty with excision of excess tissue may be indicated when conservative treatment has proven ineffective in reducing swelling but may have been successful in stabilizing disease. OBJECTIVE: To evaluate long-term results after reduction cheiloplasty in patients with macrocheilia caused by Melkersson-Rosenthal syndrome or cheilitis granulomatosa. DESIGN: Follow-up study in 7 patients with severe persisting macrocheilia, including 3 patients with Melkersson-Rosenthal syndrome and 4 patients with cheilitis granulomatosa in a stable state of disease, treated by reduction cheiloplasty at our hospital between January 1, 1987, and December 31, 2002. Preoperative and postoperative medical histories were obtained, and criteria for the success of surgical treatment were evaluated by clinical examination. Different techniques of reduction cheiloplasty are described and demonstrated in representative cases of severe macrocheilia. RESULTS: Surgical treatment in all 7 patients showed satisfying aesthetic and functional outcomes that persisted throughout follow-up (median follow-up, 6.5 years). CONCLUSIONS: Reduction cheiloplasty is an effective method to correct persistent macrocheilia and improve lip aesthetics in patients with Melkersson-Rosenthal syndrome or granulomatous cheilitis in the persistent state of disease. With careful planning, proper sequencing of treatment, and proficiency in the various surgical techniques, optimal results can be achieved.     

Granulomatous cheilitis and Crohn's disease in a 3-year-old boy

Granulomatous cheilitis and Crohn's disease are disorders of unknown etiology. There are case reports describing their coincidence and pointing out the necessity of ruling out systemic disorders once the diagnosis of granulomatous cheilitis is made. However, such reports are few and the causal association of both diseases is controversial in the literature. We report the youngest patient so far, a 3-year-old boy, who had granulomatous cheilitis and Crohn's disease simultaneously. This coincidence so early in life strongly suggests that both represent manifestations of the same disease.