Seal Finger a Case Report and Review of the Literature
Hand (N Y). 2015 Sep; ten(3): 550–554.
Solitary, adult-onset, intraosseous myofibroma of the finger: report of a case and review of literature
Yihong Ma
Department of Pathology, The University of Alabama at Birmingham, NP 3542, 619 19th St. South, Birmingham, AL 35249-7331 Usa
Cistron P. Siegal
Department of Pathology, The Academy of Alabama at Birmingham, NP 3542, 619 19th St. South, Birmingham, AL 35249-7331 Usa
Shi Wei
Department of Pathology, The University of Alabama at Birmingham, NP 3542, 619 19th St. South, Birmingham, AL 35249-7331 Usa
Abstract
Myofibroma is a rare benign tumour of myofibroblastic origin. It typically occurs in the peel and subcutaneous tissues of the caput and cervix in infants and young children as multicentric lesions known as infantile myofibromatosis. Intraosseous myofibromas are very rare and are typically destructive lesions that predominantly affect craniofacial bones in the setting of myofibromatosis. Solitary, intraosseous myofibromas in adults are exceedingly rare. Herein, nosotros report a myofibroma involving the eye phalanx of the right alphabetize finger in a 58-year-one-time man who presented with a pathologic fracture. Twelve other cases of adult-onset, intraosseous myofibroma were compiled from the English language literature and integrated with this report.
Keyword: Myofibroma, Intraosseous, Developed-onset, Solitary
Background
Myofibroma is a rare benign neoplasm of myofibroblastic origin. It typically occurs in the skin and subcutaneous tissues of the caput and cervix in infants and young children as multicentric lesions known every bit infantile myofibromatosis [1], although other anatomic sites may also be rarely affected, including bone and visceral organs. Histologically, the tumor typically exhibits a well-circumscribed, multinodular, biphasic growth pattern characterized by cellular regions consisting of plump spindled cells arranged in a hemangiopericytoma-like mode alternating with less cellular areas composed of more than mature myofibroblasts with abundant extracellular matrix. The skeletal involvement is predominantly limited to the craniofacial bones, chiefly in the setting of myofibromatosis [2–4]. Solitary, adult-onset, intraosseous myofibroma is exceedingly rare, with only a handful of case reports. These lesions, one time once again, more often than not occur in gnathic basic. Herein, we report the first example of solitary, adult-onset, intraosseous myofibroma involving the bones of easily and feet. Twelve other cases of adult-onset, intraosseous myofibroma from the English language literature were integrated with this report.
Example study
The patient, a 58-twelvemonth-former right-handed man with no significant past medical history, injured his right index finger while mowing his lawn and presented to an emergency room. Conventional radiographs demonstrated a lytic lesion in the center phalanx of the correct alphabetize finger with cortical disruption indicating a non-displaced fracture (Fig.1). He was referred to the authors' establishment for definitive management. He denied fever, chills, and unintentional weight loss.
Conventional radiographs demonstrate a well-demarcated lytic lesion with sclerotic rim and a non-displaced fracture
The patient underwent an open biopsy and curettage of the os tumor followed by grafting. The curetted specimen was composed of fragmented tan-pink soft tissue. Histopathologic examination (Fig.2a–c) of the lesion revealed a spindle cell tumour with a multinodular growth blueprint. The lesion exhibited alternating hypercellular and hypocellular zones. While the latter independent plump spindle cells with an eosinophilic cytoplasm along with prominent dystrophic calcifications. The cellular areas were composed of a mixture of spindled and round to polygonal cells arranged in a hemangiopericytoma-like growth pattern. In that location was mild nuclear atypia in areas. Mitotic figures were non discernable. No necrosis was noted. The lesional cells were immunoreactive for shine muscle actin (Fig.2nd) only negative for S-100 protein, h-caldesmon, CD34, and wide-spectrum cytokeratin cocktail (information not shown). Thus, the pathologic diagnosis of myofibroma was rendered. The postoperative grade was uneventful until 1 year later on when he noticed a swelling at the aforementioned location. A recurrence was suspected. However, the patient denied further surgical intervention.
Histologic characteristics of myofibroma. The lesion exhibits a multinodular growth pattern with alternating hypercellular and hypocellular areas and dystrophic calcification. Note the opposite zonation with the so-chosen "myoid nodules" in the center (a, ×xl). The cellular areas are composed of a mixture of spindled and circular/polygonal cells arranged in a hemangiopericytoma-like growth design (b, ×100). The myoid cells have banal nuclei and prominent cytoplasmic eosinophilia (c, ×400). The lesional cells are immunoreactive for smooth musculus action (d, ×100)
Discussion
Infantile myofibromatosis, formerly known as congenital generalized fibromatosis, was first described by Stout in 1954 [i]. Dermis and subcutis of caput and cervix regions are the most mutual locations although other anatomic sites may likewise be rarely afflicted, including bone and visceral organs. A subset of lone and multiple forms of infantile myofibromatosis are familial, with an autosomal-dominant mode of inheritance and variable penetrance. The term of myofibroma was introduced for acquired lesions by Smith et al. in 1989 [4]. The skeletal interest is predominantly limited to the craniofacial bones. Oudijk et al. analyzed 114 cases of alone, multifocal, and generalized myofibromas. Eight of 95 (8.4 %) solitary lesions were intraosseous, including iii in mandible and two in parietal bone, with others involving clavicle, scapula, and tibia, respectively. Of note, all of the eight patients with intraosseous myofibroma were younger than 25 years of historic period, of which 6 were younger than 15 years [3]. Inwards et al. reviewed 14 cases of solitary infantile myofibromatosis of bone and found all but ane involving craniofacial bones, with the latter occurring in the tibia. While the age ranged 6 months to 16 years, most of the tumors (71 %) were in patients aged 2 years or younger [2].
Upon review of the English language literature, a total of 12 acquired/developed-onset (age 15 or older) solitary, intraosseous myofibromas with detailed clinicopathologic characteristics were found and are summarized in Tabular arrayi. There were eight men and five women in this cohort including the current case, with an age range of 15 to 58 years. These lesions once more predominantly involved craniofacial bones (54 %, 7/13), exclusively affecting mandibles (northward = 6). Other sites of bone interest included clavicles, spines, and phalanx. All patients had solitary lesions. Compared to their soft tissue counterpart, intraosseous lesions are insidious when they are small. Patients may present relatively late until unfortunate fractures occur (as in the electric current example) or until pregnant bone expansion causing deformity and astringent pain. On imaging, the tumors were well-demarcated, radiolucent lesions with variable marginal sclerosis, with or without pathologic fracture. The differential diagnosis is broad and includes other benign lesions such as nerve sheath tumor, fibrous dysplasia, enchondroma, hemangioma, aneurysmal bone cyst, and an infectious process. When they announced as an ill-defined radiolucency, more aggressive lesions such equally fibrous histiocytoma, fibrosarcoma, Rosai-Dorfman disease, Langerhan cell histiocytosis, desmoid tumor and metastasis are also in the differential diagnostic consideration. Those involving appendicular bones, although extremely rare, tend to occur in the metaphysis, some demonstrating cortical expansion and thus may return a differential diagnosis of metaphyseal cortical defect or chondromyxoid fibroma.
Tabular array 1
Summary of adult-onset, intraosseous myofibromas
| Case | Author/year | Age/sex | Site | Presentation | Size (cm) | Imaging | Treatment | Issue (follow-up years) |
|---|---|---|---|---|---|---|---|---|
| 1 | Asirvatham/1994 [8] | eighteen/Yard | Cervical spine (C2) | Local hurting | N/A | Lytic lesion with fracture | Curettage and grafting | NED (four) |
| 2 | Imaizumi/1999 [9] | 15/G | Clavicle | Local pain | 2.5 | Lucent, well-confining lesion with slight expansion of the cortex | Resection and grafting | NED (4.4) |
| three | Oliver/2003 [10] | 34/F | Mandible | Incidental finding | 2.5 | Excision | North/A | |
| four | Sedghizadeh/2004 [eleven] | 20/M | Mandible | Local pain, swelling and difficulty chewing | N/A | Large radiolucent lesion with fine internal septation and scalloped margins | Resection and reconstruction | NED (0.9) |
| five | Bodkin/2005 [12] | 17/F | Temporal bone, extending into the sphenoid | Slowly growing mass, little local pain | 5 | Expansile lesion with non-sclerotic peripheral bony rims | Excision and cranioplasty | NED (ane.iv) |
| 6 | Konishi/2007 [13] | 33/M | Lumbar spine (L1 pedicle) | Severe local pain | Lytic lesion involving both corpus and lamina with a sclerotic rim | Laminectomy | NED (0.4) | |
| 7 | Biglioli/2010 [14] | 17/M | Mandible | Incidental finding | one | Lytic lesion | Excision | Due north/A |
| 8 | Ramadorai/2010 [15] | 32/F | Mandible | Painful swelling | 4.2 | Well defined expansile lytic lesion with irregular resorption of buccal cortex | Excision and reconstruction | N/A |
| 9 | Oudijk/2012 [iii] | 24/M | Mandible | N/A | 3 | North/A | Excision | N/A |
| 10 | Oudijk/2012 [3] | xv/F | Clavicle | North/A | 2.half dozen | North/A | Excision | N/A |
| 11 | Brierley/2012 [16] | 43/F | Mandible | Swelling | one | Well-demarcated, unilocular radiolucency | Curettage | North/A |
| 12 | Sundaravel/2013 [17] | xvi/M | Mandible | Swelling | 2.5 | Lytic, destroying the buccal cortical plate | Wide excision | NED (3) |
| 13 | Electric current instance | 58/Thou | Middle phalanx of alphabetize finger | Pathologic fracture | 1.five | Lytic lesion with a non-displaced fracture | Curettage and grafting | Recurrence (one) |
Histologic recognition of this entity in bone is critical since the intraosseous lesions are extremely rare, and their lytic growth pattern is difficult to be distinguished from other lesions radiographically. The microscopic appearance of bony lesions is not much different from its soft tissue analogue, which typically shows a multinodular, biphasic growth design with alternating hypercellular and hypocellular regions, although the two components may be present in variable proportions. The night-stained, cellular regions consist of densely packed circular to spindle-shaped myofibroblasts with scant eosinophilic cytoplasm and indistinct cell borders. The light-stained, less cellular areas are composed of myofibroblasts with abundant extracellular matrix that often refers to equally "myoid nodules." A perivascular growth pattern is oftentimes recognizable in the cellular areas surrounding the myoid lobules and, in fact, these lesions were initially thought to prevarication on a histologic continuum with the other tumor types having a perivascular myoid differentiation, including myopericytoma and glomangiopericytoma. The distinction between them is sometimes arbitrary due to considerable overlap. Stromal hyalinization and calcifications may be present and occasionally dominant the lesion thus complicating the diagnosis. The tumor cells are invariably bland and mitotic figures are rare to absent. The overwhelming majority of myofibromas are immunoreactive for smooth muscle actin simply lack expression of other smooth musculus markers (h-caldesmon and desmin), S-100 poly peptide, and CD34 [3, 5, 6].
The histologic differential diagnosis for bony tumors largely includes smooth muscle tumors, nerve sheath tumors, solitary fibrous tumor/hemangiopericytoma, benign fibrous histiocytoma, desmoid tumor, and perivascular myoid lesions [one, ii]. Leiomyomas typically have a more homogeneously fascicular growth pattern, with longer spindle cells with cigar-shaped nuclei and brightly eosinophilic cytoplasm, and strongly express other smooth muscle markers such as h-caldesmon and desmin. The biphasic growth pattern of myofibroma may closely resemble a schwannoma. However, the latter characteristically demonstrate fibrillary, cellular areas oft arranged in a palisading fashion (Antoni A) admixed with a seemingly distinct loose textured pattern with frequent microcysts (Antoni B). These lesions invariably testify stiff and lengthened expression of Due south-100 poly peptide. Lone fibrous tumor/hemangiopericytoma typically exhibits a patternless architecture with a staghorn branching vascular pattern and perivascular hyalinization. The tumor cells are characteristically immunoreactive with CD34. Benign gristly histiocytoma can occur in any historic period and may affect whatsoever os. The lesional cells are typically bundled in a storiform pattern intermixed with variable amount of foam cells and multinucleated giant cells and stain positively with gene XIIIa. Other perivascular myoid lesions usually lack the archetype biphasic advent and myoid nodules of myofibroma. Myopericytoma may also arise in bone and rarely causes oncogenous osteomalacia [7]. Historically, myopericytomas can be separated from myofibroma past their immunoprofiles—the former stains positively with smoothen muscle actin and h-caldesmon in dissimilarity to myofibroma, which typically shows no or minimal h-caldesmon immunoreactivity. However, it is at present considered past the WHO Working Group that myopericytoma course a morphologic spectrum with myofibroma as well as a number of other entities, including infantile hemangiopericytoma, angioleiomyoma, and glomus tumor [5]. Desmoid tumor is easily excluded by immunophenotyping.
A minor subset of patients with early-onset infantile myofibromatosis with multiorgan involvement died of affliction, while adult-onset, intraosseous myofibromas are benign and rarely recur following surgical removal of the tumor, similar to their soft tissue counterpart.
In summary, we nowadays an extraordinary rare case of adult-onset, alone, intraosseous myofibroma involving the middle phalanx of the index finger. To our knowledge, this was the 5th adult instance of myofibroma arising in non-craniofacial bones and the first reported instance in the basic of easily and feet. Review of literature revealed that the adult-onset, intraosseous myofibromas carry distinctly dissimilar from the early on-onset tumors, like to their soft tissue counterpart in adults. Thus, awareness of this uncommon tumor in the adult patients is essential to forestall mistaken diagnosis and subsequent extensive workup.
Conflict of interest
Yihong Ma declares that she has no conflict of interest.
Factor P. Siegal declares that he has no conflict of interest.
Shi Wei declares that he has no conflict of interest.
Argument of human and animal rights
In that location is no violation of human being and fauna rights.
Statement of informed consent
Informed consent was obtained.
References
1. Stout AP. Juvenile fibromatoses. Cancer. 1954;vii:953–978. doi: 10.1002/1097-0142(195409)seven:v<953::AID-CNCR2820070520>3.0.CO;2-W. [PubMed] [CrossRef] [Google Scholar]
2. Inwards CY, Unni KK, Beabout JW, et al. Alone congenital fibromatosis (infantile myofibromatosis) of bone. Am J Surg Pathol. 1991;15:935–941. doi: ten.1097/00000478-199110000-00003. [PubMed] [CrossRef] [Google Scholar]
3. Oudijk Fifty, den Bakker MA, Hop WC, et al. Lonely, multifocal and generalized myofibromas: clinicopathological and immunohistochemical features of 114 cases. Histopathology. 2012;60:E1–xi. doi: x.1111/j.1365-2559.2012.04221.ten. [PubMed] [CrossRef] [Google Scholar]
4. Smith KJ, Skelton HG, Barrett TL, et al. Cutaneous myofibroma. Mod Pathol. 1989;2:603–609. [PubMed] [Google Scholar]
five. Fletcher CDM, World Wellness Arrangement., International Agency for Enquiry on Cancer. WHO classification of tumours of soft tissue and bone. 4th ed. IARC Press: Lyon, 2013
half-dozen. Goldblum JR, Folpe AL, Weiss SW, et al. Enzinger and Weiss's soft tissue tumors. vi. Philadelphia, PA: Saunders/Elsevier; 2014. [Google Scholar]
7. Brunschweiler B, Guedj North, Lenoir T, et al. Oncogenous osteomalacia and myopericytoma of the thoracic spine: a instance report. Spine. 2009;34:E857–860. doi: 10.1097/BRS.0b013e3181b780e9. [PubMed] [CrossRef] [Google Scholar]
8. Asirvatham R, Moreau PG, Antonius JI. Solitary infantile myofibromatosis of axis. A case report. Spine. 1994;nineteen:80–82. doi: 10.1097/00007632-199401000-00018. [PubMed] [CrossRef] [Google Scholar]
9. Imaizumi S, Ogose A, Hotta T, et al. Alone infantile myofibromatosis involving the clavicle. Skeletal Radiol. 1999;28:473–476. doi: 10.1007/s002560050550. [PubMed] [CrossRef] [Google Scholar]
10. Oliver RJ, Coulthard P, Carre C, et al. Solitary adult myofibroma of the mandible simulating an odontogenic cyst. Oral Oncol. 2003;39:626–629. doi: ten.1016/S1368-8375(03)00042-3. [PubMed] [CrossRef] [Google Scholar]
11. Sedghizadeh PP, Allen CM, Kalmar JR, et al. Solitary central myofibroma presenting in the gnathic region. Ann Diagn Pathol. 2004;viii:284–289. doi: ten.1016/j.anndiagpath.2004.07.005. [PubMed] [CrossRef] [Google Scholar]
12. Bodkin PA, Choksey MS, Fagan J. Lone calvarial myofibroma presenting in boyhood. Br J Neurosurg. 2005;19:420–424. doi: ten.1080/02688690500390300. [PubMed] [CrossRef] [Google Scholar]
13. Konishi E, Mazaki T, Urata Y, et al. Solitary myofibroma of the lumbar vertebra: adult example. Skeletal Radiol. 2007;36(Suppl 1):S86–ninety. doi: 10.1007/s00256-006-0132-2. [PubMed] [CrossRef] [Google Scholar]
14. Biglioli F, Colombo V, Valassina D, et al. Miniretromandibular access for mandibular condyle biopsies. Minerva Stomatol. 2010;59:603–609. [PubMed] [Google Scholar]
15. Ramadorai A, Rajsekaran A, Narayanan V. A case written report of lonely, intraosseous, developed-onset myofibroma of the mandible. J Maxillofac Oral Surg. 2010;nine:280–283. doi: 10.1007/s12663-010-0073-0. [PMC free article] [PubMed] [CrossRef] [Google Scholar]
16. Brierley DJ, Khurram SA, Speight PM. Alone myofibroma of the developed mandible: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;115:e40–43. doi: 10.1016/j.oooo.2012.05.012. [PubMed] [CrossRef] [Google Scholar]
17. Sundaravel South, Anuthama K, Prasad H, et al. Intraosseous myofibroma of mandible: a rarity of jaws: with clinical, radiological, histopathological and immunohistochemical features. J Oral Maxillofac Pathol. 2013;17:121–125. doi: 10.4103/0973-029X.110703. [PMC free article] [PubMed] [CrossRef] [Google Scholar]
Articles from Hand (New York, N.Y.) are provided here courtesy of American Clan for Paw Surgery
Source: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4551629/
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