Solitary Orbital Infantile Myofibroma: A Case Report and Review of the Literature

Persaud, Tarek O.; Nik, Narieman A.; Keating, Robert F.; Boyajian, Michael J.; Przygodzki, Ronald; Nemi, Ajit; Miller, Michelle S.

doi:10.1016/j.jaapos.2005.11.012

Cited by 17 publications

(20 citation statements)

References 7 publications

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“…A review of the English literature based on a MEDLINE search from 1966 to 2010 was performed and all cases of infantile myofibroma involving the orbit specifically were included in the review, the majority of which were single case reports [4][5][6][7][8][9][10][11][12][13][14][15][16][17][18]. Clinical series of ''head and neck infantile myofibromatosis'' were selected if critical information about orbit lesions was included.…”

Section: Methodsmentioning

confidence: 99%

“…Specifically, if only isolated disease is present, there is a chance of excellent clinical outcome with spontaneous involution [1,6,8,11,13,14,17,18,25]. In contrast, patients who have visceral involvement may experience significant morbidity and mortality.…”

Section: Pathologic Featuresmentioning

confidence: 99%

“…While fibromatoses [3] may occur in any region of the body, many occur in the head and neck, usually involving the superficial soft tissues. Within the head and neck, infantile myofibromatosis, although a very rare lesion, seems to show a tendency to develop around the orbit [1][2][3][4][5][6][7] in the few cases reported in the English literature [4][5][6][7][8][9][10][11][12][13][14][15][16][17][18]. The rarity of this tumor poses a diagnostic challenge that may lead to a misdiagnosis and subsequent inappropriate patient management.…”

Section: Introductionmentioning

confidence: 99%

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Orbital Infantile Myofibroma: a Case Report and Clinicopathologic Review of 24 Cases from the Literature

Mynatt

Feldman²,

Thompson

2011

Head and Neck Pathol

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Isolated orbital infantile myofibroma are rare tumors in the head and neck. The mass-like clinical presentation and variable histologic features result in frequent misdiagnosis and potentially inappropriate clinical management. There are only a few reported cases in the English literature. Twenty-four patients with orbital infantile myofibroma or myofibromatosis were compiled from the English literature (Medline 1960-2011) and integrated with this case report. The patients included 14 males and 10 females, aged newborn to 10 years (mean, 34.8 months), who presented with a painless mass in the infra- or supraorbital regions, usually increasing in size andassociated with exophthalmos (n = 5). Females were on average older than their male counterparts (38.9 vs. 31.9 months, respectively; P = 0.71). The tumors were twice as frequent on the left (n = 16) than right (n = 8). Patients experienced symptoms for an average of 2.7 months before clinical presentation. The tumors involved the bone (n = 17) or the soft tissues (n = 7) of the orbit, with extension into the nasal or oral cavity (n = 3). The mean size was 3.0 cm, with a statistically significant difference between males and females (mean: 3.9 vs. 1.82; P = 0.0047), but without any differences based on age at presentation (P = 0.25), duration of symptoms (P = 0.66), or bone or soft tissue involvement (P = 0.51). Grossly, all tumors were well-circumscribed, firm to rubbery, homogenous, and white-grey. Histologically, the tumors were biphasic, showing whorled and nodular areas of fusiform cells with extracellular collagen, mixed with a population of small, primitive-appearing, darkly staining cells. Necrosis was not present, but mitoses could be seen. Tumors with immunohistochemistry performed showed strong and diffuse smooth muscle actin and vimentin immunoreactivity, but were negative with muscle specific actin, desmin, MYOD1, myogenin, S100 protein, GFAP, keratin, CD31, 34, Factor VIIIR-Ag, and CD45RB. The principle histologic differential diagnosis includes juvenile hyaline fibromatosis, fibrous hamartoma of infancy, fibromatosis coli, leiomyoma, infantile hemangiopericytoma, infantile fibrosarcoma, Ewing sarcoma/primitive neuroectodermal tumor, and lymphoma. All patients were managed with surgery. Recurrences developed in two patients at 4 and 6 months, respectively. Follow-up data was available on all but two patients (n = 22). These patients were either alive without evidence of disease (n = 18), alive but with disease (n = 3), or had died unrelated to this disease (i.e., neuroblastoma, n = 1). Orbital infantile myofibroma is a rare tumor, presenting in infancy as an enlarging mass of the orbit, with characteristic histomorphologic and immunophenotypic features. Orbital disease is usually isolated rather than part of systemic disease, and shows an excellent long-term prognosis, making appropriate separation from other conditions important.

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Section: Methodsmentioning

confidence: 99%

Section: Pathologic Featuresmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Orbital Infantile Myofibroma: a Case Report and Clinicopathologic Review of 24 Cases from the Literature

Mynatt

Feldman²,

Thompson

2011

Head and Neck Pathol

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“…41 Routine postoperative care must include assessment for hemodynamic stability, fluid balance, and neurologic assessment, including pain, as well as close monitoring for infection. It is important to remember that especially in the presence of visceral tumors, the infant's vital signs are a good indicator of cardiorespiratory compromise.…”

Section: Nursing Care and Family Supportmentioning

confidence: 99%

Infantile Myofibroma

Schurr

Moulsdale²

2008

Advances in Neonatal Care

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This article presents a case report of a 37-week gestational age (GA) female infant (CK) whose first ultrasound at 35 weeks' GA revealed polyhydramnios, fetal ascites, and a possible diaphragmatic hernia. At birth, CK had a grossly distended abdomen, prominent abdominal veins, hepatomegaly, bounding femoral pulses, and generalized edema. Initial imaging identified an absent ductus venosus, absent segment of the inferior vena cava (IVC), and prominent superior vena cava to the right atrium. A computed tomography (CT) scan showed a mass contiguous with the liver causing compression of the IVC. Biopsy confirmed infantile myofibromatosis (IM), an uncommon soft tissue neoplasm that may present at birth or in early infancy. Although rare, this neoplasm is the most common fibrous tumor of infancy. The case of CK was unusual because the solitary IM lesion was in an atypical location; a solitary lesion is not commonly found in the viscera, and solitary lesions are predominant in males. Although lesions are often benign, visceral involvement is associated with high mortality. The cause is unknown, although familial cases have been reported. This article describes the key features of IM, possible treatment options, nursing care, and prognosis for infants with the disease.

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“…The prognosis of solitary myofibroma is excellent, and complete excision is advised (Chung & Enzinger 1981;Persaud et al 2006). This diagnosis should especially be considered in a child when encountering a painless solitary orbital tumour progressing over months.…”

mentioning

confidence: 99%

In vivo evaluation of photoreceptor mosaic in type 2 idiopathic macular telangiectasia using adaptive optics

Massamba

Querques

Lamory

et al. 2010

Acta Ophthalmologica

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Solitary Orbital Infantile Myofibroma: A Case Report and Review of the Literature

Cited by 17 publications

References 7 publications

Orbital Infantile Myofibroma: a Case Report and Clinicopathologic Review of 24 Cases from the Literature

Orbital Infantile Myofibroma: a Case Report and Clinicopathologic Review of 24 Cases from the Literature

Infantile Myofibroma

In vivo evaluation of photoreceptor mosaic in type 2 idiopathic macular telangiectasia using adaptive optics

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