Yonago Acta medica 2003;46:77–81

Multiple Myeloma Presenting Symptoms in the Oral andMaxillofacial Region
Masami Shibata, Isamu Kodani, Rieko Doi, Kazuko Takubo, Kazunori Kidani,
Hiroatsu Sakai, Masayuki Sonoda and Kazuo Ryoke
Divisin of Oral and Maxillofacial Biopathological Surgery, Department of Medicine of Sensoryand Motor Organs, School of Medicine, Tottori University Faculty of Medicine, Yonago 6838503 Japan
Four cases of multiple myeloma presenting symptoms in the oral and maxillofacial region
are reported. The main complaints in 2 patients were macroglossia and dysphagia. We
doubted these cases were amyloidosis, but examinations performed for possible amyloidosis
led us to a diagnosis of multiple myeloma. The other 2 patients presented signs in themandible. One of them had a mass-type lesion in the gingiva which upon X-ray revealed
an osteolytic lesion in the mandible and multiple “punched-out” osteolytic lesions in thecranial bones. The other showed only a diffuse non-homogeneous radiolucent lesion anddental ankylosis. Medical history and biochemical and hematologic findings would be
helpful in diagnosing multiple myeloma. Here, we report our summarization of these 4cases.
Key words: amyloidosis; dysphagia; macroglossia; multiple myeloma

Amyloidosis as a complication has been reported in
6% to 15% of patients with multiple myeloma(Raubenheimer et al., 1986). This article reports 4
patients with multiple myeloma presenting symptoms in the oral and maxillofacial region.

Multiple myeloma is a malignant disease characterized by multifocal proliferation of atypical plasma
cells and, in most patients, by the presence in theserum of monoclonal gamma globulins and/or theirsubunits, often referred to as “M” or myeloma proteins (Lambertenghi-deliliers et al., 1988). Oral andmaxillofacial manifestations as an initial sign orsymptom of multiple myeloma are rare (Lee et al.,1996). The common clinical signs and symptoms
of multiple myeloma include pain in the bone, fatigue, anemia and infectious diseases (Lee et al.,
1996; Keung et al., 1998). Radiography frequently
shows the typical “punched-out” osteolytic lesions.
Amyloidosis is a disease characterized by thedeposition of an abnormal protein in a variety oforgans. The infiltration can be local or systematic.

Patient Report

Patient 1
A 55-year-old woman was referred to our hospital
because of submental swelling and tongue stiffness
on August 30, 1985. The symptoms were first
noted by the patient 5 months prior to the referral,and progressed steadily since then. First physicalAbbreviation: Ig, immunoglobulin

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M. Shibata et al.

Congo red staining of the muscle fibers revealed amyloid deposits. Upon the tentative
diagnosis of amyloidosis of the tongue, further laboratory examinations were performed. The urine was positive for Bence-Jones
protein. Immunoelectrophoresis of the serum showed a monoclonal IgA band with a κlight chain. Bone marrow aspiration revealed abundant plasma cells. These data
helped to provide a final diagnosis of amyloidosis complicated by multiple myeloma.
The patient was then referred to a hematologist for chemotherapy with melphalan andpredonisolone. Swelling of the tongue wasFig. 1. Tongue showing macroglossia and indentations caused
reduced temporarily. But 6 months later, aby the teeth (Patient 1).
partial glossectomy was performed because
the patient complained of difficulty withspeaking, eating and swallowing. In theexamination showed a mass in the submental refollowing
months there was no continuous enlargegion. Intraoral examination revealed a firm diffuse
ment of the tongue. Twenty-nine months after theswelling of the tongue. The patient’s medical hissurgical intervention, the patient died of heart faitory included arthralgia. She did not return forlure.
medical follow-up, and no definite diagnosis wasmade. But 1 month later, she returned to our hospital because of rapid growth in tongue swelling.
Patient 2Upon intraoral examination, the tongue was signifiAn 81-year-old man visited our hospital after fallcantly enlarged, firm, nodular and crenated (Fig. 1).
ing off of a bridge on September 27, 1996. Upon
The tongue was subjected to incisional biopsy:
intraoral examination, the lower right 2nd premolar
histological examination revealed a hyperplastic
and 2nd molar were invaded with caries. Panoramepithelium and atrophic muscle fibers. Amyloid
ic radiography showed an osteolytic lesion in thedeposits were distributed around the blood vessels.

Fig. 2. Panoramic view showing osteolytic lesions on the right side of the mandibular body (Patient 2).

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Oral multiple myeloma

Fig. 3. Amyloid deposits in the tongue muscle (Patient 3) (Congo red stain, original magnification × 100).

right mandibular body (Fig. 2). The medical historyof this patient included herpes zoster in 1991, anemia and multiple myeloma treated with melphalan
and predonisolone since 1995, and pneumonia andfracture of the lumber vertebra in 1996. Biochemical and hematologic findings revealed anemia andelevation in serum IgG. We extracted his lower
right 2nd premolar and 2nd molar, and observed
during curettage that the jawbone was rough. The
histological examination of the excised tissue revealed abundant atypical plasma cells. These results
are consistent with a diagnosis of multiple myeloma.

The above-described results were compatible withthe diagnosis of amyloidosis secondary to multiple
myeloma. Because of her advanced age, active
treatment against multiple myeloma was notperformed.

Patient 4
A 66-year-old man visited our hospital on May 18,
1984, because of gingival swelling and bleeding in
the lower right molar area for 1 month. Upon intraoral examination, we found 2 soft tissue masses
located in the lower right 1st molar area (38 × 31 ×28 mm) and on the lower left 3rd molar area (21 ×12 × 12 mm). The masses were elastic, hard andpediculate (Fig. 4). Panoramic radiography showed
an osteolytic lesion in the right mandibular body.
The patient’s medical history revealed hypertension. The mass was examined by an incisional biopsy: histologically, pleomorphic plasma cells
were diffusely infiltrating. On the basis of thepathologic and clinical findings, the patient wasdiagnosed with multiple myeloma, and was subjected to hematologic screening and a radiographic
survey. Skeletal radiographic survey of the skull
revealed multiple “punched-out” osteolytic lesions(Fig. 5). Immunoelectrophoresis of the serum

Patient 3
An 83-year-old woman visited our hospital on April6, 1994, because of tongue stiffness and dysphagia
lasting for about 1 month. The patient’s medical
history included aspiration pneumonia. On theintraoral examination, we observed a firm nodular
tongue with a diffuse swelling, and performed an
incisional biopsy of the tongue. Congo red stainingof the tissue revealed amyloid deposits in the muscle fibers (Fig. 3). Bence-Jones protein was detected in the urine. Serum protein immunoelectrophoresis revealed elevation in serum IgG with a κ chain.
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