Osseous mass in a maxillary sinus of an adult male from the 16th–17th-century Spain: Differential diagnosis

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Abstract

Objective

To undertake a differential diagnosis of a large mass found in the left maxillary sinus of a cranium dated to the 16th–17th-century, and to expand knowledge of the diagnosis of osseous tissue formation in osteoarchaeological studies.

Material

A cranium recovered from the cemetery of San Salvador de Palat de Rey church, León (Spain).

Methods

Macroscopic analysis, CT scanning.

Results

Macroscopic analysis indicated that the individual was probably a male over 30 years old with an ossified mass in the left maxillary sinus, measuring 24 × 19 × 24 mm, occupying approximately 27 % of the maxillary antrum. Computed tomography revealed a well-demarcated radiolucent unilocular mass with some radiopaque areas, with no communication with the alveoli of the premolars or molars. No erosive lesions or signs of inflammation were found.

Conclusions

Neither the macroscopic, nor the radiological characteristics are compatible with inflammatory or malignant pathology, favoring a diagnosis of ossifying fibroma.

Significance

This case adds to the few reported cases in the osteoarchaeological literature, especially since there is limited relevant reference data to assist diagnosis. The CT scans and 3D reconstruction presented here facilitate differential diagnosis in future paleopathological studies.

Limitations

Destructive methods were not authorized.

Suggestions for further research

In the future, micro-CT analysis, which was not performed in the current study, may add new and valuable information.

Introduction

The maxillary sinus is the largest of the four paranasal sinuses and, being anatomically adjacent to the dentate region of the maxilla, is a frequent source of pathological modification. Calcifications or formation of new bone are characteristic of lesions of the maxillary sinus and include osteosarcoma, chondrosarcoma, as well as calcifying and osteoblastic tumors. However, the site of origin is not necessarily the epicenter of the lesion (Farman and Nortjé, 2007). The maxillary sinus constitutes a path of least resistance for the growth of other maxillary lesions, such as cysts and benign neoplasms. Large benign tumors and cysts might be present without resulting in clinically perceptible maxillofacial expansion (Neville et al., 2016).

Osseous masses in the maxillary sinus have rarely been described in the osteoarchaeological literature. Although some bone tumors or tumor-like lesions have been reported in the maxillofacial region in archaeological skeletal remains (Assis et al., 2018), only three cases have been described within the maxillary sinus (Colard et al., 2008; Silva and Wasterlain, 2010; Nicklisch et al., 2018). However, it is believed that a similar range of prevalence of bone tumors affected past and present populations (Marques et al., 2018). The lower frequencies reported for past populations may be due to three main reasons: first, the frequency of bone tumors may have been lower due to the lower average life expectancy up to the 20th century (Nicklisch et al., 2018); second, not all neoplastic diseases leave traces on or in the bone, therefore missed by paleopathologists (Marques et al., 2018); and third, pathological conditions in the osteoarchaeological literature may be more difficult to differentially diagnose (Marques et al., 2018).

The scarcity of similar cases in the paleopathological literature, and the fact that such osseous masses represent a diagnostic challenge owing to the absence of histopathological analysis, makes differential diagnosis based on macroscopic and radiological characteristics relevant. This paper aims to undertake differential diagnosis of an osseous mass found within the left maxillary sinus of an adult male cranium from the cemetery of the San Salvador de Palat de Rey church in León, northwestern Spain (Fig. 1), and to assist with diagnosis in osteoarchaeological studies.

Section snippets

Archeological context

The San Salvador de Palat de Rey church is located in the historic center of the city of León, northwestern Spain (Fig. 1). This church is considered the oldest in the city. It was built by King Ramiro II “El Grande” (931/32-951 AD) in the second quarter of the 10th century, and was integrated into a monastic complex for his daughter Elvira next to the Royal Palace (Flórez, 1756). In 1215, being part of one of the most important way-stations on the pilgrimage route of Saint James of

Biological profile

The cranium PRII5 is that of a 30+-year-old male of European Ancestry (Affiliation = 1; p-value = 0.70; Accuracy = 0.94; Positive Predictive Value = 0.90). Male sex was obtained by both the morphological analysis and the discriminant function analysis. In fact, the latter indicated a 90 % probability of this cranium being that of a male.

Oral pathology

The maxillae were relatively well preserved (Fig. 1, Fig. 2). Twelve teeth were lost ante-mortem, leaving the alveolar sockets in different stages of

Discussion

Establishing a diagnosis in paleopathology, although important, is a complex procedure. The key features used to narrow down the etiological possibilities for the lesion under investigation were its macroscopic characteristics, anatomical distribution, radiographic features, as well as the individual sex, age and ancestry. The modified Istanbul Terminological Framework (Appleby et al., 2015) was adopted to aid reporting of the differential diagnosis.

Conclusions

Although differential diagnosis of the paleopathological lesions described in this study is limited, the macroscopic and radiological features of the reported osseous mass are highly consistent with a diagnosis of ossifying fibroma. During life, the León individual may have presented facial signs associated with the expansion of the tumor occupying the left maxillary sinus, such as swelling, unilateral proptosis with diplopia, persistent nasal obstruction, rhinorrhea and epiphora, as well as

Declaration of Competing Interest

The authors state that they do not have any conflict of interest to declare.

Acknowledgements

The authors thank the Centro de Investigação em Antropologia e Saúde (CIAS), Medical Image Unit - Centro Hospitalar e Universitário de Coimbra, and Erasmus + Program. Laura González-Garrido thanks Fernando Miguel Hernández and Museo de León for the access to the archaeological documentation, and Dr. Luis Caro Dobón and Dra. Humildad Rodríguez Otero for their support. Sofia N. Wasterlain was financed by national funds from the FCT - Fundação para a Ciência e Tecnologia, project grant reference

References (59)

  • N.B. Sayan et al.

    Peripheral osteoma of the oral and maxillofacial region: a study of 35 new cases

    J. Oral Maxillofac. Surg.

    (2002)
  • P.M. Speight et al.

    Maxillofacial fibro-osseous lesions

    Curr. Diagn. Pathol.

    (2006)
  • M. Traversari et al.

    Multi-analytic study of a probable case of fibrous dysplasia (FD) from Certosa monumental cemetery (Bologna, Italy)

    Int. J. Paleopathol.

    (2019)
  • M. Ulmansky et al.

    Benign cementoblastoma. A review and five new cases

    Oral Surg. Oral Med. Oral Pathol. Oral Radiol.

    (1994)
  • J.N. Waldron et al.

    Carcinoma of the maxillary antrum: a retrospective analysis of 110 cases

    Radiother. Oncol.

    (2000)
  • T.K. Bala et al.

    Cemento-ossifying fibroma of the mandible. A clinicopathological report

    Saudi Med. J.

    (2017)
  • A. Barberi et al.

    Bilateral cemento-ossifying fibroma of the maxillary sinus

    Br. J. Radiol.

    (2003)
  • L.T. Barbian et al.

    Healing following cranial trauma

    J. Forensic Sci.

    (2008)
  • J.E. Buikstra et al.

    Standards for Data Collection From Human Skeletal Remains

    (1994)
  • A.B. Burke et al.

    Fibrous dysplasia of bone: craniofacial and dental implications

    Oral Dis.

    (2017)
  • T. Colard et al.

    A paleopathological case of a right maxilla’s cemento-ossifying fibroma

    Int. J. Osteoarchaeol.

    (2008)
  • A.S. Dadhich et al.

    Cementoblastoma of posterior maxilla involving the maxillary sinus

    Ann. Maxillofac. Surg.

    (2015)
  • G. Dias et al.

    ‘Abscess cavity’ – a misnomer

    Int. J. Osteoarchaeol.

    (1997)
  • P.M. Dubal et al.

    Squamous cell carcinoma of the maxillary sinus: a population‐based analysis

    Laryngoscopes

    (2016)
  • S.K. El-Mofty et al.

    Ossifying fibroma

  • R. Eversole et al.

    Benign fibro-osseous lesions of the craniofacial complex: a review

    Head Neck Pathol.

    (2008)
  • A.G. Farman et al.

    Pathological conditions affectiong the maxillary sinus

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