Osteolytic mandible presenting as an initial manifestation of an adult acute lymphoblastic leukaemia

Abstract

A case of adult acute lymphoblastic leukaemia is reported. A 35-year-old male presented with an osteolytic lesion of the mandible. There was no definitive involvement in other craniofacial bones. A panoramic radiograph taken 4 months previously showed no bony involvement. A complete blood count showed a slightly decreased red blood cell count, but normal white blood cell count, white blood cell differential count and platelet count. Routine chemistry revealed hypercalcemia with an increased level of parathyroid hormone-related protein. Histopathological examination of bone marrow biopsy confirmed the diagnosis of acute lymphoblastic leukaemia.

Generally, osteolytic lesions of the jaws are initially detected by dental professionals. The aetiology is usually attributed to infections of dental or periodontal origin, but this hard tissue change may also represent a manifestation of systemic disease. In some hematologic disorders, osteoclast activating factors are generated that result in extensive and rapid bone destruction. The purpose of this report is to document an unusual manifestation of an adult acute lymphoblastic leukaemia (ALL).

Case report

A 35-year-old Korean male, with no known history of systemic disease, was referred for evaluation of the jaws. Four months previously, the patient had visited a local dental clinic, presenting with dull pain on the right posterior mandible. The dentist extracted the lower right first molar. There had been no excessive bleeding before or after the extraction. Two months after the extraction, the patient complained of mobility of the lower anterior and left posterior teeth. A resin-wire splint on the lower anterior teeth and extraction of the lower left second molar was carried out by a local oral surgeon. Complete blood count (CBC) and routine chemistry were normal at this time.

At the initial assessment in the authors’ department, the patient was well-oriented, but complained of fatigue. He had a mild fever (37.8 °C), but other vital signs were normal. Physical examination of the head and neck showed blepharoptosis of the left eye and left third and sixth nerve palsy. There was no evidence of dyspnoea or dysphagia. Intraoral examination revealed no petechia or any sign of infection on the oral mucosa. Previous extraction sites (lower right first molar and lower left second molar) were undergoing normal healing. A panoramic radiograph revealed an osteolytic lesion on the left mandibular body and ascending ramus area with severe vertical bone loss on the lower left posterior mandible ( Fig. 1 ). Computed tomography (CT) images showed an osteolytic lesion on the entire left half of the mandible with multiple enlarged lymph nodes in the left submandibular space ( Fig. 2 ). Acute osteomyelitis of the mandible was considered as a possible differential diagnosis, but discarded as there had been no possible origin of odontogenic infection in the oral cavity. Osteosarcoma of the jaws was also considered, but excluded as there was absence of localized pain, swelling and radiopaque lesion on radiographic images. A tentative diagnosis of hematologic malignancy was made as neurological symptoms were present. The patient was admitted for systemic evaluation. CBC showed a slight decrease in red blood cell (RBC) count (3.93 × 10 ⁶/μl), but normal white blood cell (WBC) count (10.39 × 10 ³/μl), WBC differential count and platelet count (261 × 10 ³/μl). Routine chemistry revealed an extremely high calcium level (18.2 mg/dl). The patient was transferred to the Department of Nephrology for evaluation and correction of hypercalcemia. Immediate hemodialysis and intravenous infusion of Miacalcic inj (Salcatonin, Novartis) 500 IU/ml was performed to correct the hypercalcemia. Twelve hours later, the calcium level dropped to 12.7 mg/dl. The same therapy was given on the second day of hospitalization, but the calcium level rose again to 18.0 mg/dl on the third day. Another intravenous infusion of Salcatonin was given, with oral administration of Actonel (Risedronate sodium, Handok) 35 mg. Despite these measures, the calcium level did not decrease below 15.0 mg/dl until the sixth day. On the sixth day, the patient suddenly developed pneumonia, acute respiratory distress syndrome and was sent to the intensive care unit for ventilator care after oral intubation. On the seventh day, multiple organ failure developed with unstable blood pressure and the patient died. Positron emission tomography (PET) CT taken on the fifth day revealed increased fluorodeoxyglucose uptake in the whole skeleton ( Fig. 3 ). Bone marrow biopsy performed on the fourth day later confirmed the diagnosis of ALL, Burkitt type.