Multiple Calcifying Hyperplastic Dental Follicle (MCHDF): A Case Report

Jamshidi, Shokoofeh; Zargaran, Massoumeh; Mohtasham, Nooshin

doi:10.5681/joddd.2013.028

Cited by 8 publications

(4 citation statements)

References 7 publications

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“…Regional odontodysplasia, initially documented by Hitchin in 1934, exhibits cementum-like and psammomatous calcification, enveloped by a whorled fibrous stroma referred to as CWN in this investigation [23]. Multiple calcifying hyperplastic dental follicles represent a rare condition characterized by numerous unerupted teeth with substantial calcifications and remnants of odontogenic epithelium within their enlarged dental follicles, often accompanied by odontodysplasia [6][7][8][9][10][11]14]. Histologically, all cases demonstrated similarities, showcasing calcified materials with a whorled or spiral structure akin to CWN.…”

Section: Discussionmentioning

confidence: 76%

“…Multiple calcifying hyperplastic dental follicles (MCHDFs) represent a rare pathological condition that was initially described by Sandler et al in 1988 [6]. The term MCHDF was coined by Gardner DG and Radden B in 1995 [7], and subsequently, numerous authors reported similar cases using this nomenclature [8][9][10][11][12][13][14]. These cases share common histological features, displaying small droplets of calcified material with a whorled pattern and varying amounts of odontogenic epithelial islands within fibrous or myxofibrous tissue.…”

Section: Introductionmentioning

confidence: 99%

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Developmental Anomalies in Human Teeth: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting with DSP, Nestin, and HES1

Hasegawa,

Shimada,

Ochiai

et al. 2024

JDB

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Hyperplastic dental follicles (HDFs) represent odontogenic hamartomatous lesions originating from the pericoronal tissues and are often associated with impacted or embedded teeth. These lesions may occasionally feature unique calcifying bodies, known as calcifying whorled nodules (CWNs), characterized by stromal cells arranged in a whorled or spiral fashion. CWNs are typically observed in multiple calcifying hyperplastic dental follicles or regional odontodysplasia. In our study, we examined 40 cases of HDFs, including nine instances with characteristics of CWNs, referred to as calcifying hyperplastic dental follicles (CHDFs), which are infrequently accompanied by odontodysplasia. The median ages of the HDFs and CHDFs were 16 (ranging from 3 to 66) and 15 (ranging from 11 to 50) years, respectively. The lower third molars were the most frequently affected by HDSFs and CHDFs, followed by the upper canines. A histological examination was conducted on all 40 cases, with an immunohistochemical analysis performed on 21 of them. Among the cases with CWN, nine affected a single embedded tooth, with one exception. CWNs exhibited diverse calcifications featuring sparse or entirely deposited psammoma bodies, and some displayed dentinoid formation. Immunohistochemically, the stromal cells of HDFs were frequently positive for CD56 and nestin. By contrast, CWNs were negative for CD56 but positive for nestin as well as hairy and enhancer split 1 (HES1), with a few dentin sialoprotein (DSP)-positive calcified bodies. Our results revealed that hamartomatous CHDFs can impact multiple and single-embedded teeth. CWNs composed of nestin and HES1-positive ectomesenchymal cells demonstrated the potential to differentiate into odontoblasts and contribute to dentin matrix formation under the influence of HES1. This study is the first report documenting odontoblastic differentiation in HDFs. The rare occurrence of HDFs and CHDFs contributes to limited comprehension. To prevent misdiagnosis, a better understanding of these conditions is necessary.

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Section: Discussionmentioning

confidence: 76%

Section: Introductionmentioning

confidence: 99%

Developmental Anomalies in Human Teeth: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting with DSP, Nestin, and HES1

Hasegawa,

Shimada,

Ochiai

et al. 2024

JDB

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“…MCHDFs are rare, and their etiology is still unclear ( 158 ); they are clinically defined by multiple unerupted teeth and large DFs ( 159 ). Radiographically, these follicles are observed as radiolucency surrounding the crown of the unerupted tooth and may also exhibit radiopaque lesions within the inner part of the DF ( 160 , 161 ). The histological features of this condition include extensive cemento-like calcification and the presence of residual odontogenic epithelium within the fibrous connective tissue matrix ( 162 ).…”

Section: Syndromes and Genetic Disordersmentioning

confidence: 99%

Abnormal dental follicle cells: A crucial determinant in tooth eruption disorders (Review)

Chen,

Ying,

et al. 2024

Mol Med Rep

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The dental follicle (DF) plays an indispensable role in tooth eruption by regulating bone remodeling through their influence on osteoblast and osteoclast activity. The process of tooth eruption involves a series of intricate regulatory mechanisms and signaling pathways. Disruption of the parathyroid hormone-related protein (PTHrP) in the PTHrP-PTHrP receptor signaling pathway inhibits osteoclast differentiation by DF cells (DFCs), thus resulting in obstructed tooth eruption. Furthermore, parathyroid hormone receptor-1 mutations are linked to primary tooth eruption failure. Additionally, the Wnt/β-catenin, TGF-β, bone morphogenetic protein and Hedgehog signaling pathways have crucial roles in DFC involvement in tooth eruption. DFC signal loss or alteration inhibits osteoclast differentiation, affects osteoblast and cementoblast differentiation, and suppresses DFC proliferation, thus resulting in failed tooth eruptions. Abnormal tooth eruption is also associated with a range of systemic syndromes and genetic diseases, predominantly resulting from pathogenic gene mutations. Among these conditions, the following disorders arise due to genetic mutations that disrupt DFCs and impede proper tooth eruption: Cleidocranial dysplasia associated with Runt-related gene 2 gene mutations; osteosclerosis caused by CLCN7 gene mutations; mucopolysaccharidosis type VI resulting from arylsulfatase B gene mutations; enamel renal syndrome due to FAM20A gene mutations; and dentin dysplasia caused by mutations in the VPS4B gene. In addition, regional odontodysplasia and multiple calcific hyperplastic DFs are involved in tooth eruption failure; however, they are not related to gene mutations. The specific mechanism for this effect requires further investigation. To the best of our knowledge, previous reviews have not comprehensively summarized the syndromes associated with DF abnormalities manifesting as abnormal tooth eruption. Therefore, the present review aims to consolidate the current knowledge on DFC signaling pathways implicated in abnormal tooth eruption, and their association with disorders of tooth eruption in genetic diseases and syndromes, thereby providing a valuable reference for future related research.

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“…Multiple calcifying hyperplastic dental follicles (MCHDF) represent a rare pathological condition initially described by Sandler et al in 1988 [6]. The term MCHDF was coined by Gardner DG and Radden B in 1995 [7], and subsequently, numerous authors reported similar cases using this nomenclature [8][9][10][11][12][13][14]. These cases share common histological features, displaying small droplets of calcified material with a whorled pattern and varying amounts of odontogenic epithelial islands Disclaimer/Publisher's Note: The statements, opinions, and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s).…”

Section: Introductionmentioning

confidence: 99%

Human Tooth Developmental Anomaly: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting DSP, Nestin, and HES1

Hasegawa,

Shimada,

Ochiai

et al. 2023

Preprint

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Hyperplastic dental follicles (HDFs) represent odontogenic hamartomatous lesions originating from pericoronal tissues and are often associated with impacted or embedded teeth. These lesions may occasionally feature unique calcifying bodies, known as calcifying whorled nodules (CWN), characterized by stromal cells arranged in a whorled or spiral fashion. CWNs are typically observed in multiple calcifying hyperplastic dental follicles or regional odontodysplasia. In our study, we examined 40 cases of HDFs, including nine instances with characteristics of CWNs, which were infrequently accompanied by odontodysplasia. Histological examination was conducted on all 40 cases, with immunohistochemical analysis performed on 21 of them. Among the cases with CWN, nine affected a single embedded tooth, with one exception. CWNs exhibited diverse calcifications featuring sparse or entirely deposited psammoma bodies, and some displayed dentinoid formation. Immunohistochemically, stromal cells of HDFs were frequently positive for CD56 and nestin. In contrast, CWNs were negative for CD56 but positive for nestin and HES1, with a few DSP-positive calcified bodies. Our results revealed that hamartomatous calcifying HFDs can impact multiple and single-embedded teeth. CWNs composed of nestin and HES1-positive ectomesenchymal cells demonstrated the potential to differentiate into odontoblasts and contribute to dentin matrix formation under the influence of HES1. This study is the first report documenting odontoblastic differentiation in HDFs.

show abstract

Multiple Calcifying Hyperplastic Dental Follicle (MCHDF): A Case Report

Cited by 8 publications

References 7 publications

Developmental Anomalies in Human Teeth: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting with DSP, Nestin, and HES1

Developmental Anomalies in Human Teeth: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting with DSP, Nestin, and HES1

Abnormal dental follicle cells: A crucial determinant in tooth eruption disorders (Review)

Human Tooth Developmental Anomaly: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting DSP, Nestin, and HES1

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