Yeo2018 Article InfiltratingAngiolipomaOfTheFo

You might also like

Download as pdf or txt
Download as pdf or txt
You are on page 1of 7

See discussions, stats, and author profiles for this publication at: https://www.researchgate.

net/publication/322370667

Infiltrating angiolipoma of the foot: magnetic resonance imaging features and


review of the literature

Article in Skeletal Radiology · January 2018


DOI: 10.1007/s00256-017-2870-8

CITATIONS READS

6 80

4 authors, including:

Euidong Yeo
VHS Medical Center
36 PUBLICATIONS 351 CITATIONS

SEE PROFILE

All content following this page was uploaded by Euidong Yeo on 15 June 2020.

The user has requested enhancement of the downloaded file.


Skeletal Radiology (2018) 47:859–864
https://doi.org/10.1007/s00256-017-2870-8

CASE REPORT

Infiltrating angiolipoma of the foot: magnetic resonance imaging


features and review of the literature
Eui Dong Yeo 1 & Bo Mi Chung 2 & Eun Ju Kim 3 & Wan Tae Kim 2

Received: 8 November 2017 / Revised: 25 December 2017 / Accepted: 27 December 2017 / Published online: 10 January 2018
# ISS 2018

Abstract
Angiolipoma is a benign soft tissue tumor with two subtypes: non-infiltrating and infiltrating. Although histologically benign,
infiltrating angiolipoma can invade surrounding structures. The foot is a very rare location for angiolipoma, with only four cases
reported in the English literature, including one infiltrating type. Here, we report a very rare case of infiltrating angiolipoma of the
foot with magnetic resonance imaging (MRI) and ultrasonography (US) findings. A 7-year-old boy presented with a slowly
growing foot mass. MRI showed an unencapsulated mass involving the third web space extending to the foot dorsum and sole.
The mass was isointense to subcutaneous fat and was mixed with internal T1 low-signal-intensity enhancing areas. On US, we
observed a heterogeneously hypoechoic mass with internal vascularity. Imaging and clinical features of angiolipoma and the
radiologic differential diagnoses of a fat-containing mass in the pediatric foot are reviewed here. When there is an ill-defined foot
mass with a fat component and variable enhancing portions in a child, infiltrating angiolipoma should be included in the
differential diagnosis along with other fat-containing tumors.

Keywords Angiolipoma . Infiltrating angiolipoma . MRI . Foot mass

Introduction ultrasonography (US) findings [5]. Here, we report a very rare


case of an infiltrating angiolipoma with demonstration of MRI
Angiolipoma is a benign soft tissue tumor with two subtypes: and US features in a 7-year-old boy who presented with slow-
non-infiltrating and infiltrating. Although angiolipomas are a ly growing foot mass. We reviewed the imaging and clinical
histologically benign soft tissue tumor, infiltrating-type features of angiolipoma and the radiologic differential diagno-
angiolipomas are partially or entirely unencapsulated and ses of fat-containing masses in the pediatric foot. When poorly
can infiltrate the surrounding soft tissues or bones [1]. The demarcated soft tissue tumor of foot with macroscopic fat on
foot is an unusual location for angiolipoma with only four MRI is encountered, infiltrating angiolipoma could be consid-
previously reported cases [2–5]. To our knowledge, there ered preoperatively.
has been only one case report of infiltrating angiolipoma of
the foot, without magnetic resonance imaging (MRI) or
Case report
* Bo Mi Chung A 7-year-old boy presented with a mass at the third web space
bom1086@naver.com
of the right foot. The mass was movable, soft, and non-painful
on palpation. Reddish and bluish skin discolorations were
1
Department of Orthopaedic Surgery, Veterans Health Service seen at the plantar and dorsal aspect. No negative impact on
Medical Center, 53, Jinhwangdo-ro 61-gil, Gangdong-gu,
Seoul 05368, Republic of Korea
function or neurologic abnormality was noted. The mass had
2
been noted at the time of birth and had been growing slowly.
Department of Radiology, Veterans Health Service Medical Center,
53, Jinhwangdo-ro 61-gil, Gangdong-gu, Seoul 05368, Republic of
Past medical and family history and general physical exami-
Korea nation were negative.
3
Department of Pathology, Veterans Health Service Medical Center,
Radiograph of the foot demonstrated a soft tissue mass at
53, Jinhwangdo-ro 61-gil, Gangdong-gu, Seoul 05368, Republic of the third web space with a curly deformity of the fourth toe.
Korea No mineralization or bone destruction was noted (Fig. 1).
860 Skeletal Radiol (2018) 47:859–864

performed; however, based on imaging and operative and his-


tologic findings, the final diagnosis was infiltrating
angiolipoma. The patient’s postoperative course was uncom-
plicated, and there was no local recurrence after 6-month fol-
low-up.

Discussion

Angiolipoma is a benign soft tissue tumor that was established


as a distinct entity in 1960 by Howard and Helwig [6]. It is a
well or poorly encapsulated mass and consists of mature adi-
pocytes and varying degrees of angiomatous proliferation.
The presence of fibrinous microthrombi is a distinctive feature
that differentiates this from other lipomas [4, 7]. In 1966,
Gonzlez-Crussi et al. distinguished between non-infiltrating
and infiltrating types [8]. The non-infiltrating type is more
common, and it typically presents as multiple, small
(<2 cm), slowly growing, painful lumps that are rarely asso-
Fig. 1 Plain radiograph of the foot demonstrated a soft tissue mass ciated with overlying skin discoloration. These are generally
(asterisk) at the third web space with curly deformity of the fourth toe. seen in pubescent patients [7, 9]. The infiltrating type is par-
No mineralization or bone destruction was noted tially or entirely unencapsulated and can infiltrate surrounding
tissues, leading to muscular pain and neural deficits [6].
MRI revealed a mass involving the third web space and adja- Angiolipomas occur most frequently in the forearm,
cent dorsal and plantar aspects of the forefoot at the subcuta- followed by the trunk and upper extremity [8]. Angiolipoma
neous layer, measuring 3.5 cm in the maximal dimension. A of the foot is a very rare entity, and only four case reports have
coronal T1-weighted image revealed a fatty mass isointense to been published in the English literature (Table 1) [2–5]. Two
subcutaneous fat, with hypointense tubular, tortuous struc- out of four previously reported cases were non-infiltrating
tures (Fig. 2a). The fatty component showed signal suppres- type angiolipoma, one case was infiltrating type, and the type
sion on a T2-weighted, fat-suppressed image and showed no was not specified in one case report. None of the masses
enhancement. The non-fatty component demonstrated slightly caused functional or neurologic disability, but two of four
high signal intensity (SI) on a T2-weighted, fat-suppressed patients complained of difficulty in putting on shoes or walk-
image and showed enhancement on a contrast-enhanced T1- ing. Three out of four cases had grown slowly [2–5]. Our
weighted image with fat suppression (Fig. 2b, c). It was a patient had a painless mass that had slowly grown since birth,
poorly demarcated mass, and the boundary between the fatty which is in accordance with previous reports. Skin change is
component of the mass and subcutaneous fat was not definite. reported to be uncommon, but our patient showed skin discol-
There was no apparent bone infiltration. On US, the lesion oration at the plantar and dorsal aspects of the foot. This was
was a heterogeneous echoic mass with ill-defined margin. probably due to the superficial location and predominant vas-
Internal vascularity was seen on color Doppler US (Fig. 3a, b). cular component. Due to its tendency for local recurrence, the
During surgery, a 6-cm web space incision was made. After preferred treatment for infiltrating angiolipoma is complete
incision, a lobulated and yellowish lesion was seen mixed excision. In cases of recurrence or inadequate resection, adju-
with dark-red structures, which thought to be vessels. The vant radiotherapy is considered [3, 10]. However, in cases
mass had an ill-defined margin and was not bordered by the where adequate excision would result in compromised func-
deep intermetatarsal ligament. Partial excision was performed, tion or morbidity, treatments need to be individualized
preserving neurovascular structures. Microscopically, the [11–13]. In our case, partial excision was performed, preserv-
mass was comprised of mature adipose and proliferated vas- ing neurovascular structures and tendons. There have been
cular tissue without signs of atypia in either of the two com- few reports of infiltrating foot angiolipoma, so it is difficult
ponents. There were capillary proliferations within the adi- to compare treatment choices; however, many authors report-
pose tissue, and there were areas of large vessels with blood ed a good prognosis even with subtotal removal of spinal
pools or thrombi (Fig. 4a–c). The features were considered in infiltrating angiolipomas, as these lesions are slowly growing
keeping with angiolipoma. No immunohistochemistry analy- and do not undergo malignant transformation [11–13].
sis or genetic marker study was performed. The entire margin On MRI, the fatty component of angiolipoma shows high
could not be evaluated because partial excision was SI on T1-weighted images and signal suppression on fat-
Skeletal Radiol (2018) 47:859–864 861

Fig. 2 MR coronal images. T1-


weighted (a), fat-suppressed T2-
weighted (b), and fat-suppressed
T1-weighted images with
enhancement (c) show an ill-de-
fined, infiltrative mass
involving the third web space,
foot dorsum, and sole. It is a fatty
mass isointense to subcutaneous
fat (asterisks) with internal T1
hypointense, enhancing tortuous
structures (arrowheads)

Fig. 3 Transverse images in


grayscale (a) and color Doppler
ultrasonography (b). The ill-
defined mass (arrows) was
heterogeneously hypoechoic
compared to subcutaneous fat and
had internal vascularity
862 Skeletal Radiol (2018) 47:859–864

Fig. 4 a The angiolipoma was comprised of mature adipose and tissue (asterisks) (H&E middle magnification). c Vascular proliferations
proliferated vascular tissue without signs of atypia in either of the two were filled with blood pools or thrombi (long arrows) (H&E middle
components [hematoxylin and eosin (H&E) low magnification]. b magnification)
Capillary proliferations (arrowheads) are interspersed within the adipose

suppressed images. The vascular component shows areas of tissue tumors in the pediatric population include adipocytic
low SI on T1-weighted images and high SI on fat-suppressed tumors (lipoma, angiolipoma, lipoblastoma, liposarcoma),
T2-weighted images. The ratio of these components varies fibroblastic/myofibroblastic tumors (fibrous hamartoma of in-
and is reflected by MRI features. More vascular components fancy, lipofibromatosis), and vascular tumors (involuting
lead to more prominent T1 hypointense, enhancing regions hemangioma) [9, 15–17]. Lipoma is a homogeneous fatty
seen in the mass [1, 7]. In this case, there was a rich vascular mass identical to subcutaneous fat with or without a thin,
component microscopically, which corresponded with MRI fibrous capsule or septa with low SI [9]. The substantial pro-
findings that showed predominant, tortuous T1 hypointense portion of the enhancing area in our case enabled us to rule out
areas. On US, angiolipomas are reported as hyperechoic le- a simple lipoma. Lipoblastoma and, more rarely, liposarcoma
sions compared to muscle, and flow is present in less than are usually encapsulated masses with enhancing or necrotic
25% of cases [14]. Our case showed an ill-defined mass with components, in contrast to the non-encapsulated mass in our
echogenicity slightly higher than that of muscle and lower case [9, 15]. Fibrous hamartoma of infancy presents as a poor-
than that of subcutaneous fat with internal vascularity, which ly defined subcutaneous fat-containing mass with a heteroge-
was in accordance with the literature. neous SI area of fibro-collagenous tissue and primitive mes-
Soft tissue tumors usually have a nonspecific imaging ap- enchymal cells. An organized pattern of alternating bands is
pearance, but identification of intralesional macroscopic fat characteristic, and the most frequent sites are the upper ex-
enables radiologists to narrow the differential diagnosis, tremity, upper trunk, and neck. The characteristic MRI pattern
which would be most commonly benign. Fat-containing soft and prevalent sites could be helpful for differentiation [9, 16].
Skeletal Radiol (2018) 47:859–864 863

Table 1 Summary of published case reports

Case summary Authors Type Modality Treatment

F/12 painless mass at the 1st and Gravante G et al. 2006 Not specified X-ray, MRI Wide excision Letter to editor
2nd toes, recurred and slowly
grew after wide excision
M/47 soft nodular mass at the Theodoros B Grivas et al. 2008 Non-infiltrating type X-ray, CT, MRI Marginal excision Full paper
plantar aspect of forefoot, with
painful gait. Detected 25 years prior
and slowly increased in size
F/33 painless mass at foot dorsum on Tighe C et al. 1994 Non-infiltrating type X-ray Marginal excision Full paper
the 1st and 2nd metatarsals, grew
slowly for 1 year, produced difficulty
putting on shoes
M/51 mass with ulceration distal to the Wertheimer SJ et al. 1992 Infiltrating type X-ray Wide excision Full paper
metatarsal head. Amputation state at
metatarsophalangeal joint level

In our case, the mass was a non-encapsulated, fat-containing areas containing both adipose tissue and a vascular compo-
mass in the foot, and the differential diagnoses included nent, which is consistent with angiolipoma.
lipofibromatosis and involuting hemangioma. Immunohistochemically, endothelial cells in angiolipomas
Lipofibromatosis is a heterogeneous mass with varying com- are positive for endothelial markers (CD34, CD31). Some
position of fat and fibroblastic cells that most frequently oc- authors observed an increased number of mast cells and the
curs at the hands and feet. They present as heterogeneously presence of cytokines produced by mast cells including vas-
hyperintense masses on T1-weighted image with poorly de- cular endothelial growth factor (VEGF) in immunostaining
marcated margins. There are T1-low SI septa or small nodules and suggested that it might be responsible for vascular prolif-
that show high SI on fat-suppressed T2-weighted images, eration in angiolipoma [19, 20].
though there is never a fluid signal. Enhancement is variable Infiltrating angiolipoma is a rare soft tissue tumor in the
depending on the proportion of fibroblasts [9, 17]. The en- foot, with few previously reported cases. When there is an ill-
hancing portion of our case was linear or tubular, favoring a defined foot mass with a fat component and variable enhanc-
vascular component rather than fibroblasts. Involuting hem- ing portions in a child, infiltrating angiolipoma should be in-
angiomas are ill-defined fatty masses that are often indistin- cluded in the differential diagnosis along with other fat-
guishable from adjacent subcutaneous fat, with varying containing tumors. Despite the ill-defined margin of the mass,
amounts of vascular components. The differential diagnosis detection of macroscopic fat on MRI can help narrow the
with imaging features is difficult. In addition, there is contro- differential diagnosis, suggest benign nature preoperatively,
versy about the entity of infiltrating angiolipomas and heman- and prevent aggressive and potentially disfiguring wide
giomas with fatty overgrowth. Gonzlez-Crussi et al. classified resection.
unencapsulated lipomatous lesions with a vascular component
in the skeletal muscles and deep soft tissue as infiltrating Compliance with ethical standards
angiolipomas [8]. However, Enzinger and Weiss state that
Conflict of interest The authors declare that they have no conflict of
many of the so-called infiltrating angiolipomas reported in
interest.
the literature might have been intramuscular hemangiomas
with fatty overgrowth [1]. Some authors think that there is a
continuum between the intramuscular hemangioma and the
infiltrating lipoma, with the infiltrating angiolipoma
References
representing an intermediate stage between the two [18].
However, the characteristic clinical features of the phasic
1. Arenaz Bua J, Luaces R, Lorenzo Franco F, Garcia-Rozado A,
and evolusive pattern can help. These lesions become appar- Crespo Escudero JL, Fonseca Capdevila E, et al. Angiolipoma in
ent in the first few weeks or months of life, rapidly grow until head and neck: report of two cases and review of the literature. Int J
about 1 year of age, and then slowly involute over the next Oral Maxillofac Surg. 2010;39(6):610–5.
several years [9]. The mass grew slowly in our case, which is 2. Tighe C, Lynn JA. Angiolipoma of the foot. A review of the liter-
ature and case report. J Am Podiatr Med Assoc. 1994;84(2):85–9.
not the typical clinical course of an involuting hemangioma. 3. Gravante G, Monaco A, Delogu D, Filingeri V, Esposito G. Foot
Microscopically, there were capillary proliferations inter- angiolipomas: the third case of the literature. Eur Rev Med
spersed in the adipose tissue component and encapsulated Pharmacol Sci. 2006;10(2):87–9.
864 Skeletal Radiol (2018) 47:859–864

4. Grivas TB, Savvidou OD, Psarakis SA, Liapi G, Triantafyllopoulos 14. Bang M, Kang BS, Hwang JC, Weon YC, Choi SH, Shin SH, et al.
G, Kovanis I, et al. Forefoot plantar multilobular noninfiltrating Ultrasonographic analysis of subcutaneous angiolipoma. Skelet
angiolipoma: a case report and review of the literature. World J Radiol. 2012;41(9):1055–9.
Surg Oncol. 2008;6:11. 15. Chen CW, Chang WC, Lee HS, Ko KH, Chang CC, Huang GS.
5. Wertheimer SJ, Balazsy JE. Infiltrating angiolipoma in the foot. J MRI features of lipoblastoma: differentiating from other palpable
Foot Surg. 1992;31(1):17–24. lipomatous tumor in pediatric patients. Clin Imaging. 2010;34(6):
6. Howard WR, Helwig EB. Angiolipoma. Arch Dermatol. 1960;82: 453–7.
924–31. 16. Stensby JD, Conces MR, Nacey NC. Benign fibrous hamartoma of
7. Kitagawa Y, Miyamoto M, Konno S, Makino A, Maruyama G, infancy: a case of MR imaging paralleling histologic findings.
Takai S, et al. Subcutaneous angiolipoma: magnetic resonance im- Skelet Radiol. 2014;43(11):1639–43.
aging features with histological correlation. J Nippon Med Sch. 17. Vogel D, Righi A, Kreshak J, Dei Tos AP, Merlino B, Brunocilla E,
2014;81(5):313–9. et al. Lipofibromatosis: magnetic resonance imaging features and
8. Gonzalez-Crussi F, Enneking WF, Arean VM. Infiltrating pathological correlation in three cases. Skelet Radiol. 2014;43(5):
angiolipoma. J Bone Joint Surg Am. 1966;48(6):1111–24. 633–9.
9. Sheybani EF, Eutsler EP, Navarro OM. Fat-containing soft-tissue 18. Agamanolis DP, Dasu S, Krill CE Jr. Tumors of skeletal muscle.
masses in children. Pediatr Radiol. 2016;46(13):1760–73. Hum Pathol. 1986;17(8):778–95.
10. Alvi A, Garner C, Thomas W. Angiolipoma of the head and neck. J
19. Silva-Junior GO, Picciani BL, Costa RC, Barbosa SM, Silvares
Otolaryngol. 1998;27(2):100–3.
MG, Souza RB, et al. Oral soft-tissue angiolipoma: report of two
11. Han SR, Yee GT, Choi CY, Lee CH. Infiltrating spinal angiolipoma.
cases of rare oral lipomatous lesion with emphasis on morpholog-
J Korean Neurosurg Soc. 2012;52(2):161–3.
ical and immunohistochemical features. J Oral Sci. 2013;55(1):85–
12. Gelabert-Gonzalez M, Garcia-Allut A. Spinal extradural
8.
angiolipoma: report of two cases and review of the literature. Eur
Spine J. 2009;18(3):324–35. 20. Atilgan AO, Terzi A, Agildere M, Caner H, Ozdemir BH.
13. Guzey FK, Bas NS, Ozkan N, Karabulut C, Bas SC, Turgut H. Intraosseous angiolipoma of the frontal bone with a unique loca-
Lumbar extradural infiltrating angiolipoma: a case report and re- tion: a clinical and pathological case illustration and review of the
view of 17 previously reported cases with infiltrating spinal literature. Indian J Pathol Microbiol. 2014;57(2):301–4.
angiolipomas. Spine J. 2007;7(6):739–44.

View publication stats

You might also like