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Heterotopic and idiopathic ossification in the left foot of a child


European Journal of Dermatology. Volume 19, Number 6, 637-9, November-December 2009, Correspondence

DOI : 10.1684/ejd.2009.0772


Author(s) : Keiji Sugiura, Mariko Sugiura, Kazuhiro Kurita, Toshiro Okagawa , Department of Environmental Dermatology and Allergology, Daiichi Clinic, Nittochi Nagoya Bld. 2F, 1-1 Sakae 2, Nakaku, Nagoya, 460-0008, Japan, Department of Orthopedics, Aichi Aoitori Institution for Disabled, 5-89, Nakaotai, Nishiku, Nagoya, 452-0822, Japan, Department of Rehabilitation, Aichi Aoitori Institution for Disabled, 5-89, Nakaotai, Nishiku, Nagoya, 452-0822, Japan.

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ARTICLE

Auteur(s) : Keiji Sugiura1, Mariko Sugiura1, Kazuhiro Kurita2, Toshiro Okagawa3

1Department of Environmental Dermatology and Allergology, Daiichi Clinic, Nittochi Nagoya Bld. 2F, 1-1 Sakae 2, Nakaku, Nagoya, 460-0008, Japan
2Department of Orthopedics, Aichi Aoitori Institution for Disabled, 5-89, Nakaotai, Nishiku, Nagoya, 452-0822, Japan
3Department of Rehabilitation, Aichi Aoitori Institution for Disabled, 5-89, Nakaotai, Nishiku, Nagoya, 452-0822, Japan

A 6-year-old boy with autism had developed a round shaped hard swelling (figure 1) on his left foot the age of 3 or 4. This swelling had gradually elevated and sometimes shown inflammation. Our first diagnosis was calcifying epithelioma. X-ray showed calcification on his left foot, inside his big toe (figure 1), possibly due to ossification or polydactyly of the left foot. We treated him with surgical removal under local anesthesia. Histopathological findings showed bone tissue formation (figure 1) and no cartilage tissue. Our final diagnosis was idiopathic and heterotopic ossification (HO).

Generally heterotopic ossification (HO) is present after surgical treatment [1, 2] or trauma [3, 4]. His parents said this patient had not had any surgical operations or traumas. Therefore the origin of this ossification was unknown. Because this patient had autism, he may not have been able to express the presence of any traumas very well. We do not know when his HO developed; it was possibly present --in his left foot as early as infancy. Generally, in many polydactyly cases, bone formations are like ramifications. X-ray findings showed this case to be similar to polydactyly, because it resembles the bones in an infant stage, especially like the epiphyseal line. There were no continuances to other bones and there were no arteries and veins for this HO. Three disorders are common as ossification: Fibrodysplasia ossifications progressiva (FOP), Albright hereditary osteodystrophy (AHO) and Progressive osseous heteroplasia (POH). We discuss these disorders in table 1, which is useful for differential diagnosis. Generally, patients with autism do not have any ossification, and this patient did not have any disease and syndrome except for autism. This patient had an isolated ossification. Our final diagnosis was heterotopic idiopathic ossification in the left foot. Radiotherapy is effective for HO [5, 6]. Because this patient was 6 years old, we did not use radiotherapy due to consideration of side effects. Our treatment was a surgical operation under local anesthesia for diagnosis, there were no findings of recurrence and development after 5 months. This case was uncommon based on the origin of the HO, the clinical findings, and the age of patient.
Table 1 Differential diagnosis for ossification disease

Inheritance, gene mutation

Ossification

Sex dominant

Onset

Characters and features

Limitation of mobility

Flare-up

Hormone

Calcium

Prognosis

Treatment

Fibrodysplasia ossifications progressiva (FOP)

Gene encoding the stimulatory G protein, gene encoding the BMPI recepter (ACVR1), chromosome 2

Skeletal muscle, ligaments, connective tissue, endochondral

Infancy

Torticollis, deformity of thorax and neck, joint immobilization, malformation of big toe

Severe

Severe, suddenly

Require assistance for life

No effective

Albright hereditary osteodystrophy (AHO)

Maternal or paternal inheritance, GNAS1 gene, mutation in chromosome 20, deletion in the region 2q37 of chromosome

Skin and subcutaneous

Short neck, short stature, mental retardation, round face, osteoporosis, low nasal bridge

No severe

High parathyroid hormone

Hypocalcemia

Good

Progressive osseous heteroplasia (POH)

Paternally inheritance, GNAS1 gene

Cutaneous and connective tissue

Female

Congenital and childhood

Severe

Normal

Normal

Better

No definitive

Idiopathic heterotopic ossification

None

Skin and subcutaneous

None

None

None

Normal

Normal

Good

Remove

Acknowledgements

Conflict of interest: none. Financial support: none.

References

1 Safaz I, Alaca R, Bilgiç S, Yazicioğlu K. A heterotopic ossification related to temporary placement of a skull bone flap. Case report. J Neurosurg 2008; 108: 370-1.

2 Stoltny T, Koczy B, Wawrzynek W, Miszczyk L. Heterotopic ossification in patients after total hip replacement. Ortop Traumatol Rehabil 2007; 9: 264-72.

3 Gautschi OP, Cadosch D, Bauer S, Filgueira L, Zellweger R. Heterotopic ossification-from the aetiology to the current management. Unfallchirug 2008; 111: 523-34.

4 Siemers F, Lohmeyer JA, Machens HG, Eisenbeiss W, Mailänder P. Heterotopic ossifications: a severe complication following extensive burn injury. Handchir Mikrochir Plast Chir 2007; 39: 360-3.

5 Zarzycka M, Reszke J, Windorbska W, Ziółkowska E. Radiotherapy in the prevention of heterotopic ossification after arthroplasty. Ortop Traumatol Rehabil 2002; 4: 230-6.

6 Nasr E, Nehme R, Ghanem I, Azoury F, Nasr DN, Dagher F. Role of radiotherapy in heterotopic ossification. Cancer radiother 2009; 13: 42-6.


 

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