Case Report

A 18 year old man, with the chief complaint of ulcer and pain on buccal and lingual gingiva of his upper left premolars and first molar, was diagnosed as aggressive periodontitis.

He had severe inflammation and gingival recession with focal areas of plaque and calculus not corresponding to the amount of recession. Vertical and horizontal bone loss in the region was detected in periapical radiographs. He had a positive history of alcohol consumption, smoking, sleep problems and severe fatigue since two years ago. These factors probably contributed to the pain and ulcer and made him visit a periodontologist after three weeks. He underwent routine periodontal treatment (scaling and root planning) and periodontal surgery was recommended and planned for him. An incisional biopsy was taken from the buccal gingiva of upper left first premolar under local anaesthesia and sent to the oral pathology for final diagnosis. Histological sections showed sheets of oval cells with plump coffeeseed shaped nuclei and vast cytoplasm, mixed with large numbers of eosinophils which lead to the diagnosis of Histiocytosis X.


Then the patient was sent for total body scan and evaluation of his skeleton, but the patient declined due to the temporary healing of his lesion. During the last two years, the patient has experienced periods of progression and remissions, but not full recovery. He encountered similar lesions in the posterior area of all jaw quadrants and underwent extraction of all his posterior teeth and also canines since severe bone loss and consequent severe mobility made them hopeless. Curettage was introduced and samples of soft and hard tissue from the deep tissues were sent for histopathological evaluation. Soft tissue samples showed the histopathological features of eosinophilic granuloma which was approved by S100 and CD1a positive immunohistochemical staining. Hard tissue samples taken from the treated tooth sockets illustrated only a reactive bone; hence the final diagnosis was soft tissue eosinophilic granuloma. Paraclinical evaluations like Complete Blood Cell counts (CBC) and total body scan was requested and checked.

The results were in normal range and no involvements of other bones were detected. Extraction of the involved teeth with an accurate curettage was performed. This provided a good healing result for the patient so that he was referred for prosthetic procedures afterwards. The patient has been followed up every 4 months for one year and no recurrence has been notified.
Eosinophilic granuloma (EG) is the mildest and localized form of Langerhans’- cell diseases formerly called Histiocytosis X. The term was introduced as a collective designation for a spectrum of clinicopathological disorders characterized by proliferation of histiocyte-like cells accompanied by varying numbers of eosinophils, plasma cells and multinucleated giant cells. Monostotic or polyostotic eosinophilic granuloma of bone is a solitary or multiple bone lesions without visceral involvement and it may occur over a wide age range but more than 50% of all cases are seen in 1st and 2nd decade of life. The jaws are affected in 10% to 20% of all cases. Dull pain and tenderness often accompany bone lesions. Radiographically, the lesions often appear as punched out radiolucencies without a corticated rim. Bone involvement in the mandible usually occurs in the posterior areas and a characteristic "scooped out" appearance may be evident when the superficial alveolar bone is destroyed (alveolar and mixed types of the lesion). Extensive alveolar involvement may result in "floating in air" appearance of teeth [2-3]. Thus tooth mobility and deep periodontal pockets may be found. Ulcerative or proliferative mucosal lesions with erythema and severe bleeding tendency on probing may develop if the disease breaks out of the bone. These clinical and radiographic features resemble severe aggressive periodontitis (AP), which can be ruled out by appropriate laboratory tests and histopathological evaluation.

We report a case of EG which initially was diagnosed and treated as AP.