UNIVERSIDADE ESTADUAL PAULISTA
JÚLIO DE MESQUITA FILHO”
Instituto de Ciência e Tecnologia
Campus de São José dos Campos
CASE REPORT DOI: https://doi.org/10.4322/bds.2024.e4283
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Braz Dent Sci 2024 July/Sept;27 (3): e4283
This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in
any medium, provided the original work is properly cited.
Diagnostic and therapeutic pathways of lip lesions in onco
hematology: report of a challenging pediatric case
Vias diagnósticas e terapêuticas de lesões labiais em onco-hematologia: relato de um caso pediátrico desafiador
Victória Boëchat FEYO1 , Iasminy Soares de Oliveira ROELES1 , Júlia Campos FABRI2 , Lydia Silva PROVINCIALI3 , Tereza
Cristina ESTEVES4 , Maria das Graças Afonso Miranda CHAVES1 , Gisele Maria Campos FABRI1
1 - Universidade Federal de Juiz de Fora, Faculdade de Odontologia, Juiz de Fora, MG, Brazil.
2 - Faculdade de Ciências Médicas e da Saúde, Faculdade de Medicina, Juiz de Fora, MG, Brazil.
3 - Faculdade de Ciências Médicas e da Saúde, Faculdade de Odontologia, Juiz de Fora, MG, Brazil.
4 - Universidade Federal de Juiz de Fora, Faculdade de Medicina, Juiz de Fora, MG, Brazil.
How to cite: Feyo VB, Roeles ISO, Fabri JC, Provinciali LS, Esteves TC, Chaves MGAM, et al. Diagnostic and therapeutic pathways of lip lesions
in onco hematology: report of a challenging pediatric case. Braz Dent Sci. 2024;27(3):e4283. https://doi.org/10.4322/bds.2024.e4283
ABSTRACT
Background: Oncohematologic patients have a high incidence of thrombosis, due to either their the underlying
disease or treatment. The standard management involves low molecular weight anticoagulants; however, these
agents may increase bleeding risk. Case report: A rare case of acute hemorrhagic lesion in pediatric patient
is described, emphasizing the diagnostic challenges encountered. An eight-year-old male with severe anemia,
fever, and thrombosis of the superior and inferior vena cava was admitted for diagnostic evaluation. Enoxaparin
was prescribed, and the diagnosis of anaplastic large cell lymphoma (ALCL) was conrmed. After 20 days of
anticoagulant therapy, the patient developed epistaxis followed by the formation of oral hemorrhagic edema in
the upper and lower lips. A specic protocol of oral care with multiprofessional team was implemented.The lesion
showed resolution, along with physical and emotional recovery. Conclusion: The personalized care protocol
successfully restored essential oral functions, including chewing, swallowing, digestion, taste, and speech, thereby
redening the true signicance of a smile; crucial in oncohematological patients.
KEYWORDS
Dentistry; Oncology; Oral cavity; Pediatrics; Thrombosis.
RESUMO
Contexto: Pacientes oncohematológicos tem elevada taxa de trombose devido ao tratamento ou à doença. O
tratamento preconizado para esses casos é o anticoagulante de baixo peso molecular, entretanto pode aumentar
o risco de sangramentos. Relato de caso: Descreve-se um caso raro de lesão hemorrágica aguda na infância
discutindo o desao do diagnóstico. Paciente do sexo masculino, 8 anos, internado para investigação diagnóstica.
Apresentava anemia grave associada a febre e trombose das veias cavas superior e inferior, por isso prescreveu-se
enoxaparina. O diagnóstico foi de linfoma anaplásico de grandes células. Após 20 dias da administração do
anticoagulante, o paciente desenvolveu epistaxe seguida de formação de trombo hemorrágico oral em lábio
superior e inferior. Um protocolo especíco de cuidados bucais envolveu a avaliação da equipe multiprossional.
Houve remissão da ferida com o restabelecimento físico e emocional. Conclusão: conforme protocolo individual
de atendimento houve restabelecimento das funções vitais da cavidade oral: mastigação, deglutição, digestão,
paladar, fala, resignicando o real signicado do sorriso; crucial em pacientes oncohematológicos.
PALAVRAS-CHAVE
Odontologia; Oncologia; Cavidade bucal; Pediatria; Trombose.
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Braz Dent Sci 2024 July/Sept;27 (3): e4283
Feyo VB et al.
Diagnostic and therapeutic pathways of lip lesions in onco hematology: report of a challenging pediatric case
Feyo VB et al. Diagnostic and therapeutic pathways of lip lesions in onco
hematology: report of a challenging pediatric case
INTRODUCTION
Lymphomas are classified into Hodgkin
or non-Hodgkin types, affecting B-cells or
T-cells [1]. Anaplastic large cell lymphoma (ALCL)
is a subtype characterized by the malignant
proliferation of T-cells and is classied as a non-
Hodgkin lymphoma, which can be ALK-positive
with ALK protein expression or ALK-negative
without the expression of this protein. Although
morphologically similar, these subtypes exhibit
distinct molecular characteristics that inuence
prognosis [2]. ALCL predominantly affects
pediatric patients, constituting 10-30% of all
lymphomas, with a notably higher prevalence in
males. The development of ALCL is linked to several
factors, including genetic and environmental
inuences, though precise mechanisms remain
under investigation [1,2]. Clinical manifestations
include peripheral lymphadenopathy, which can
be mediastinal, abdominal, or cervical, as well
as extranodal involvement affecting skin, bones,
soft tissues, central nervous system, and kidneys.
Thrombocytopenia, fatigue, and pallor due to
anemia may also occur [2,3]. Oral manifestations
are rare but may present as ulcerative and
hemorrhagic masses on the alveolar ridge, palate,
tongue, or lips [3]. The oral cavity manifestations
arising from oncologic treatment include
mucositis, xerostomia, opportunistic infections,
inammation and carious lesions [4,5].
ALCL treatment involves multi-agents che-
motherapy regimens, including anthracyclines.
Common protocols including CHOP (cyclophos-
phamide, doxorubicin hydrochloride, vincris-
tine sulfate and prednisone) with etoposide.
For relapsed cases, alternative therapies such as
ICE (ifosfamide, carboplatin, and etoposide) and
DHAP (dexamethasone, high dose Ara C, known
as cytarabine, and cisplatin) are used [6,7]. Long-
term follow-up is essential to monitor possible
complications from the antineoplastic therapy,
such as tumor lysis syndrome, infection, throm-
bosis, pancreatitis, secondary neoplasms, cogni-
tive dysfunction and endocrinopathy [8]. These
complications contribute to increased morbidity,
affecting the patient’s quality of life and psycho-
logical well-being during treatment [9].
One of the recurrent complications in
these patients is venous thromboembolism,
often associated with a hypercoagulable state.
In some cases, thrombocytosis is observed
prior to chemotherapy, suggesting a possible
tumor-related inuence on platelet production
and function. This raises questions about the
role of platelets in tumor development and
progression [10]. Moreover, chemotherapy
can alter coagulation factors, contributing to
a hypercoagulable state, which may induce a
prothrombotic condition [11].
Current guidelines recommend low molecular
weight heparin for the treatment and prevention
of thrombosis, as it offers a favorable prognosis.
However, due to the associated risk of bleeding,
careful monitoring is essential [12,13]. Direct oral
anticoagulants, while approved for thrombosis
management, have not been adequately studied
in pediatric oncology patients [12-14].
Given the complications of chemotherapy
on clotting factors and its repercussions on the
oral cavity, we report a rare case of an ALCL
in a pediatric patient who developed extensive
hemorrhagic lesions on the upper and lower lips
during the anticoagulant and chemotherapeutic
therapy, along with the collaborative therapeutic
approach of the dental and multidisciplinary
team.
CASE REPORT DESCRIPTION
An eight-years-old male was admitted to a
regional referral hospital in Juiz de Fora, Brazil,
for evaluation of severe anemia, fever, and
thrombosis affecting both the superior and the
inferior vena cava. Enoxaparin was initiated as the
immediate therapeutic intervention. The diagnostic
hypothesis was anaplastic large cell lymphoma, and
complementary tests were requested to conrm
this: myelogram and immunophenotyping. This
study was approved by the Ethics Committee of
the Universidade Federal de Juiz de Fora under a
protocol number 3.294.048.
Twelve days after starting the anticoagulant
therapy, the patient developed hemorrhagic
lesions on the upper and lower lip (Figure 1).
A dental team consultation revealed
hemorrhagic bluish-reddish lesions on both
lips, along with dehydration, paleness and mild
epistaxis. In addition, increased skin dehydration
from epithelial desquamation and fungal
infections at the labial commissure were observed.
The patient’s habitual biting and licking of the
lips, as reported by his caregiver, exacerbated
the epithelial desquamation. This condition
signicantly affected the patient’s overall well-
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Braz Dent Sci 2024 July/Sept;27 (3): e4283
Feyo VB et al.
Diagnostic and therapeutic pathways of lip lesions in onco hematology: report of a challenging pediatric case
Feyo VB et al. Diagnostic and therapeutic pathways of lip lesions in onco
hematology: report of a challenging pediatric case
being and led to refusal of food. After consulting
with the medical team and evaluating the oral
hemorrhagic complications, it was decided to
reduce the enoxaparin dosage from 60 mg/ml to
40 mg/ml and proceed with wound debridement.
The treatment included lip humidification
with physiological saline solution and debridement
of the thrombus under topical anesthesia with
benzocaine. Local bleeding was managed with
compression using sterile gauze and physiological
saline. Post-debridement, fungal lesions were found
beneath the bleeding area. Rigorous debridement
was followed by photodynamic therapy (Photon
Lase III - PL7336, DMC) at a 90 degrees angle to
the lip contact area (660 nm, 50 mW, 4 J / cm 2,
90 seconds) specically on the wound.
Topical nystatin was prescribed to treat fungal
infections at the labial commissure and under the
labial thrombus. In addition, alpha-tocopherol
acetate (vitamin E) was also recommended for its
antioxidant and hydrating properties.
The patient underwent the photodynamic
therapy every 48 hours (Figures 2 and 3), with
all other medications maintained. After 15 days,
the dental interventions and the anticoagulant
dosage adjustment resulted in total lesion
resolution and signicant physical and emotional
recovery (Figure 4). The patient continued to be
monitored during treatment and there was no
recurrence of the oral lesions.
DISCUSSION
This case report emphasizes the defiant-
strategy of approach of a pediatric patient
with ALCL who developed signicant orofacial
hemorrhagic complications related to anticoagu-
lant therapy. The extensive bleeding lesions on
both lips not only caused pain but also impacted
the patient’s diet and overall well-being, and even
covered up areas of fungal infection. This case
represents a rare occurrence in clinical practice
and is notably underrepresented in the scien-
tic literature, especially within the context of
pediatric oncology. The oral ndings can affect
the prognosis of severe diseases like ALCL and
emphasize the need for comprehensive orofacial
care strategies.
Figure 1 - Initial clinical appearance of the lip thrombus on first
examination. Figure 2 - Clinical aspects after three dental care sessions.
Figure 3 - Clinical aspects after six dental care sessions.
Figure 4 - Clinical aspects upon case resolution.
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Braz Dent Sci 2024 July/Sept;27 (3): e4283
Feyo VB et al.
Diagnostic and therapeutic pathways of lip lesions in onco hematology: report of a challenging pediatric case
Feyo VB et al. Diagnostic and therapeutic pathways of lip lesions in onco
hematology: report of a challenging pediatric case
The hospitalization was due to severe anemia,
fever, and thrombosis, symptoms consistent
with lymphoma-associated immunological
imbalance [2,3]. Twelve days post-anticoagulant
therapy initiation, the patient developed oral
hemorrhagic lesions and epistaxis. The literature
identifies only five comparable cases with
different etiologies, with this case uniquely linked
to anticoagulation. In contrast, the other lesions
reported in previous studies were primarily
associated with thrombus formation [15-19].
The scarcity of similar cases in ALCL patients
underscores the novelty of this presentation.
Following the oral manifestation, the
dental team’s systematic examination identied
clinical signs of epistaxis that corroborated the
diagnosis of the hemorrhagic lip lesion as possibly
related to anticoagulation [20-24]. The patient
also presented fungal infections in the labial
commissure, which can be attributed to ALCL-
related immunosuppression [4].
Treatment involved the debridement of
hemorrhagic crust and necrotic tissue with
benzocaine topical anesthesia [25] and
photodynamic therapy to improve cicatrization
and tissue regeneration [26]. In addition, topical
nystatin was prescribed for fungal infections [27],
and vitamin E for hydration and antioxidant
benets [28].
The primary limitation of this case report
lies in the scarcity of similar clinical conditions.
Nevertheless, the individualized oral care
protocol led to full resolution of the lesions,
improved patient nutrition, pain relief and overall
physical and emotional well-being. This case
highlights the importance of a multidisciplinary
approach in managing oral complications in
oncohematological patients. It also serves
as a valuable resource for care teams of the
oncohematologic patients, providing insights
into the diagnostic approach and decision-
making process when confronted with similar
complications.
CONCLUSION
This case-report highlights the therapeutic
challenges in managing oral complications among
pediatric oncology patients. An interdisciplinary
approach, thorough clinical examination, detailed
medical history,deep patient record analysis and
in-depth evaluation of complementary laboratory
exams were all crucial in addressing the patient’s
complex needs. Thus strengthening the concept of
orofacial care based on the individual rather than
disease. Evidence-based dental treatment tailored
to the patient’s specic condition signicantly
improved prognosis and restored vital oral
functions, including chewing, swallowing,
digestion, taste, and speech [4], Consequently,
this treatment redefined the significance of a
smile in oncohematological care.
Author’s Contributions
VBF, ISOR, LSP, GMCF: Conceptualization.
VBF, ISOR: Data Curation. VBF, ISOR, GMCF:
Formal Analysis. VBF, ISOR, GMCF: Investigation.
GMCF, TCE, MGAMC: Methodology, Project
Administration and Validation. ISOR, GMCF,
MGAMC, CPN, TCE: Resources. GMCF, MGAMC,
TCE: Supervision. ISOR, VBF, GMCF, CPN, JCF,
LSP: Visualization. ISOR, VBF, GMCF, JCF,
LSP, MGAMC, TCE: Writing - Original Draft
Preparation. ISOR, VBF, GMCF, CPN, JCF, LSP,
MGAMC:Writing - Review & Editing.
Conict of Interest
The authors declare no conict of interest.
Funding
None.
Regulatory Statement
This study was approved by the Ethics Com-
mittee of the Universidade Federal de Juiz de
Fora.
The approval code for this study is 3.294.048.
Acknowledgements
We are grateful to Dr. Cristina de Paula
Novaes for her invaluable contribution during the
technical phase of low-power laser application in
this particular clinical case.
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Gisele Maria Campos Fabri
(Corresponding address)
Universidade Federal de Juiz de Fora, Departamento de Clínica Odontológica,
Faculdade de Odontologia, Juiz de Fora, MG, Brazil.
E-mail: gisele.fabri@odontologia.ufjf.br
Date submitted: 2024 Feb 28
Accept submission: 2024 Sept 17