Australian Dental Journal

The official journal of the Australian Dental Association

Australian Dental Journal 2017; 62: 386–389

doi: 10.1111/adj.12525

Gingival enlargement in a pregnant woman with acute

monocytic leukaemia: a case report
Y-W Fu, H-Z Xu
Department of Stomatology, The First People’s Hospital of Lianyungang, Lianyungang, China.

ABSTRACT
The objective of the present study was to report the case of a pregnant woman with severe gingival enlargement for
3 months with undiagnosed acute leukaemia. The pregnant woman presented with anaemia and generalized gingival
enlargement. A provisional diagnosis of gingival enlargement in pregnancy was made. Twelve days after the initial treat-
ment, the patient was referred and admitted to the haematology department of a local hospital with clinical signs of
anaemia and thrombocytopenia. Blood count showed a white blood cell count of 9.68 9 109/L, with a haemoglobin
count of 64.0 g/L and a platelet count of 17 9 109/L. Bone marrow aspiration showed 94.5% monoblasts, and the mor-
phological diagnosis was acute monocytic leukaemia. One day after admission, the patient delivered a male infant by
Caesarean section. Ten days after the Caesarean section, the patient was started on a course of chemotherapy. Pul-
monary infection, hypokalaemia, and respiratory failure developed, and the patient died 23 days after the Caesarean sec-
tion. The present case shows the importance of awareness of severe gingival enlargement as an initial oral sign of acute
leukaemia.
Keywords: Acute monocytic leukaemia, gingival diseases, gingival enlargement, leukaemia, pregnancy.
Abbreviations and acronyms: AML = acute myeloid leukaemia; RT–PCR = reverse transcription polymerase chain reaction.
(Accepted for publication 23 April 2017.)

intervention can improve the chances of remission for

INTRODUCTION
Gingival enlargement is an increase in size of the gin-
giva and is a common feature of gingival disease.1 Sev-
eral systemic diseases may give rise to gingival
enlargement through different mechanisms. Acute leu-
kaemia is the most serious condition related to gingi-
val enlargement. In the meantime, hormonal changes
occurring during pregnancy are also associated with
generalized gingival enlargement.2 Thus, leukaemic
gingival enlargement in pregnancy may be misdiag-
nosed because it is infrequently seen in dental practise.
Leukaemias are cancers of the blood or bone mar-
row. Most leukaemias are either myeloid or lympho-
cytic/lymphoblastic, depending on which cells they
affect, and can be either chronic or acute.3 Leukaemia
during pregnancy is rarely encountered. The preva-
lence of pregnancy-associated leukaemia is approxi-
mately 1/10 000 pregnancies,4 and acute leukaemia is
estimated to occur in 1/75 000 pregnancies.5 Leukae-
mia often presents as a medical emergency, leaving lit-
tle time to consider the consequences of
chemotherapeutic interventions.6 Early diagnosis and
386
patients with leukaemia.
Here, we present an unusual case of acute mono-
cytic leukaemia during pregnancy initially presenting
as generalized gingival enlargement. In this case, the
failure to recognize the significance of the oral symp-
toms of leukaemia led to a delay in diagnosis. In addi-
tion, we provide a brief review of previous published
reports on the current knowledge of leukaemic gingi-
val enlargement.
CASE REPORT
A 27-year-old woman who was 8 months pregnant
presented to our Department of Stomatology with
fatigue, anaemia and gingival enlargement that had
been progressing over the past 3 months. Before visit-
ing our hospital, she had been treated with supragin-
gival scaling and systemic antibiotics at a local dental
clinic, but her gingival enlargement had not improved.
The patient was diagnosed with iron deficiency anae-
mia during her prenatal care in the second trimester.
She did not take any medication except ferrous sulfate
© 2017 Australian Dental Association

Fig. 1 Generalized gingival enlargement affecting both upper and lower
gingivae in our patient.
for the treatment of anaemia. Her physical examina-
tion revealed pale appearance and generalized gingival
enlargement (Fig. 1). The enlarged gingiva was moder-
ately firm and had a shiny surface. Necrotizing ulcera-
tive inflammatory involvement was observed in the
maxillary left posterior palatal gingiva. This area was
yellowish brown and painful. Plaque and calculus dis-
tribution were insignificant. A provisional diagnosis of
gingival enlargement in pregnancy was made. Oral
hygiene instructions were clearly explained to her. The
pockets were irrigated with a 0.1% aqueous solution of
chlorhexidine, and she was instructed to rinse the
mouth with 0.2% chlorhexidine twice daily. The
patient was advised to return the next day for further
examination of peripheral blood smear but did not
attend.
Twelve days after the initial treatment, the patient
was referred to the department of haematology of a
local hospital with clinical signs of anaemia and
thrombocytopenia. On admission, the patient was
pale and feverish, and had scattered purpura on the
extremities. No lymph nodes and no spleen were pal-
pable. Blood count showed a white blood cell count
of 9.68 9 109/L, with a haemoglobin count of 64.0 g/
L and a platelet count of 17 9 109/L. Bone marrow
aspiration showed 94.5% monoblasts (Fig. 2), and
the morphological diagnosis was made as acute mono-
cytic leukaemia, French–American–British classifica-
tion M5b. Immunophenotyping surface marker
analysis of leukaemic cells from bone marrow by flow
cytometry was positive for CD7 (17.0%), CD13
(61.8%), CD33 (99.5%), CD34 (3.5%) and CD117
(6.5%). CD56 expression was not studied. Chromoso-
mal analysis performed on bone marrow revealed a
normal 46, XX, (16) karyotype. The reverse transcrip-
tion polymerase chain reaction (RT–PCR) analysis dis-
closed the MLL–AF9 fusion gene.
After an interdisciplinary conference involving haema-
tology, maternal–foetal medicine, anaesthesiology and
© 2017 Australian Dental Association
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Leukemic gingival enlargement in pregnancy
Fig. 2 Bone marrow aspirate morphology from our patient showing a
monomorphic population of monoblasts (Wright–Giemsa stain, original
magnification 91000).
intensive care medicine, the decision was made to per-
form an urgent Caesarean section using general anaes-
thesia. One day after admission, the patient delivered an
infant by Caesarean section. The infant was male,
3100 g, with Apgar scores of 8 at 1 min and 9 at 5 min,
respectively. The infant was discharged home in good
condition on day 7 of life. During the Caesarean section,
the patient received 4 units of packed red cells and
1 unit of platelets. Ten days after the Caesarean, the
patient was started on a course of chemotherapy. During
her hospital stay, the patient was instructed to rinse her
mouth with 0.2% chlorhexidine twice daily. Pulmonary
infection, hypokalaemia and respiratory failure devel-
oped, and the patient died 23 days after the Caesarean
section. The baby is now 24 months of age and in good
general condition.
DISCUSSION
Gingival enlargement may be suggestive of underlying
systemic disease, such as acute leukaemia, as in the
case presented herein. Leukaemic gingival enlargement
in pregnancy is easily misdiagnosed because it is rarely
encountered in dental practise. There are some hor-
monal and physiological changes during pregnancy in
a woman’s body. The changes may make the diagno-
sis of leukaemia more challenging. The diagnosis may
be missed or delayed since non-specific symptoms of
leukaemia such as weakness, fatigue, anaemia and
gingival enlargement are sometimes attributed to preg-
nancy.2,4 Examination of bone marrow aspirate sam-
ples is currently considered a reliable method for the
diagnosis of leukaemia. Similar to all other biopsies
performed under local anaesthesia, a bone marrow
biopsy can be safely carried out during pregnancy
without harming the foetus.4
Leukaemic cells can infiltrate the gingiva and result
in gingival enlargement. Gingival enlargement in
patients with leukaemia has been described in several
387

Y-W Fu and H-Z Xu
subtypes of acute myeloid leukaemia (AML),7–19
chronic myeloid leukaemia,20 acute lymphocytic leu-
kaemia21 and chronic lymphocytic leukaemia.22,23
Furthermore, acute myelomonocytic leukaemia (M4)
and acute monocytic leukaemia (M5) are reportedly
more prone to gingival enlargement.8 A study of 1076
patients with leukaemia demonstrated that 38 (3.6%)
of the 1049 dentulous patients had leukaemic gingival
enlargement, with the highest incidence in patients
with acute monocytic leukaemia (66.67%), followed
by those with acute myelomonocytic leukaemia
(18.52%) and those with acute myelocytic leukaemia
(3.68%).24 The case reported here supports these find-
ings and represents the M5 subtype of AML.
Clinically, acute monocytic leukaemia is associated
with a high frequency of extramedullary involvement
(most notably the skin, gum and central nervous sys-
tem).25 Extramedullary involvement is associated with
the presence of CD56 antigen on blast cells and kary-
otypic abnormalities involving chromosome 11, in
particular abnormalities of 11q23 involving the mixed
lineage leukaemia (MLL) gene.15,20 In our case, RT–
PCR analysis disclosed the MLL–AF9 fusion gene.
Moreover, acute monocytic leukaemia usually has a
poorer prognosis than other subtypes of AML.
Patients with acute monocytic leukaemia are supposed
to receive careful clinical evaluation and early inten-
sive care unit admission to afford a higher chance of
recovery by using non-invasive diagnostic and thera-
peutic strategies.26
The coexistence of leukaemia and pregnancy is
uncommon. There is still no standard approach for
the current clinical dilemma.5 The majority of the leu-
kaemias diagnosed during pregnancy are acute. Of
these acute leukaemias, more than two-thirds are
myeloid.5,27 Acute leukaemia during pregnancy should
be managed by a multidisciplinary team that includes
haematologists, obstetricians and neonatologists.
Treatment should be started as soon as possible after
the diagnosis of acute leukaemia is made. Treatment
delays may compromise maternal outcome.28 Gener-
ally, leukaemic gingival enlargement resolves com-
pletely or partly with effective chemotherapy.10,24
Differential diagnosis of leukaemic gingival enlarge-
ment includes inflammatory enlargement, drug-induced
gingival enlargement, pregnancy gingival enlargement,
neoplastic enlargement and false enlargement. In the
present case, the lack of response to conventional treat-
ment should serve as a warning that the initial diagnosis
was probably incorrect. Leukaemia gingival enlarge-
ment is characterized by progressive proliferation of
fibrous connective tissue causing enlargement of the
interdental papilla, and marginal and entire attached
gingiva. However, pregnancy-related gingival enlarge-
ment may be localized or generalized, and is usually
noted on the marginal gingiva and interdental papilla.2
388
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Pre-existing periodontal disease can exaggerate the
leukaemic infiltration and worsen the gingival enlarge-
ment.29 In the present case, plaque and calculus distri-
bution were insignificant in the initial oral
examination. Gingival enlargement in the patient
might have been due to leukaemic infiltration and
hormonal changes during pregnancy. Considering the
dramatic gingival enlargement, it appears that leukae-
mic infiltration might have been the major cause of
the marked gingival enlargement. Biopsy can be used
to confirm leukaemic cell infiltration.9,12,17–20,24 In
patients with diagnosed leukaemia, the findings of gin-
gival biopsy may be of interest, but their benefit to
the patient is insufficient. In our case, the gingival
biopsy was not performed.
As gingival enlargement is one of the earlier signs
of AML, early diagnosis and intervention can improve
a patient’s chances of remission. Given the rapid pro-
gression and aggressive behaviours of the disease, den-
tists are responsible for early diagnosis in a certain
portion of patients with AML. In the meantime, medi-
cal consultation must be essential in periodontal and
dental treatment in such cases.
ACKNOWLEDGEMENTS
No financial support was received for this work and
was self-funded by the authors. Dr Fu wrote the paper.
Professor Xu reviewed and edited the manuscript. We
thank Yan-Hui Li for assistance in preparation of the
manuscript and Lei Jiang for the provision of material.
DISCLOSURE
The authors declare that they have no conflict of
interest.
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Address for correspondence:
Dr Yong-Wei Fu
Department of Stomatology
The First People’s Hospital of Lianyungang
182 North Tongguan Road
Haizhou District
Lianyungang 222002, Jiangsu
China
Email: fuyongweivip@sina.com
389