Skip to main content
Top
Published in: Pediatric Surgery International 3/2020

01-03-2020 | Original Article

Follow-up outcomes of pediatric patients who underwent surgical resection for lipoblastomas or lipoblastomatosis: a single-institution experience with a systematic review and meta-analysis

Authors: Dyda Dao, Anna J. Najor, Philip Y. Sun, Forough Farrokhyar, Christopher R. Moir, Michael B. Ishitani

Published in: Pediatric Surgery International | Issue 3/2020

Login to get access

Abstract

Purpose

The objective of this study was to examine the long-term outcomes of pediatric patients who underwent surgical resection for lipoblastoma and lipoblastomatosis (LB/LBM).

Methods

A single-center retrospective study of pediatric patients with LB/LBMs seen between 1991 and 2015 was conducted. A systematic review, including studies published prior to late August 2018, was performed. Using a random effect meta-analysis, pooled weighted proportions and unadjusted odds ratios (OR) with 95% confidence intervals (CI) were calculated.

Results

The retrospective study included 16 patients, while the systematic review included 19 published studies consisting of 381 patients. Among 329 (82%) patients with follow-up information, the pooled recurrence rate was 16.8% (95% CI 10.9–23.5%; I2 = 59%). The reported time to recurrence ranged from < 1 to 8 years. Recurrence risk was greater for incomplete (n = 34) than complete resection (n = 150): OR 11.4 (95% CI 3.0–43.6; I2 = 43%). LBMs (n = 35) had a greater recurrence risk than LBs (n = 116): OR 5.5 (95% CI 1.9–15.9; I2 = 0%). Recurrences were higher for studies with approximately ≥ 3 years of follow-up versus studies with < 3 years of follow-up.

Conclusion

Recurrences are more likely to occur with LBMs and/or incomplete resection. Follow-up beyond 3–5 years should be considered given that the recurrence risk appears to be greater in the long-term.
Appendix
Available only for authorised users
Literature
1.
go back to reference Shen LY, Amin SM, Chamlin SL, Mancini AJ (2017) Varied presentations of pediatric lipoblastoma: case series and review of the literature. Pediatr Dermatol 34:180–186PubMed Shen LY, Amin SM, Chamlin SL, Mancini AJ (2017) Varied presentations of pediatric lipoblastoma: case series and review of the literature. Pediatr Dermatol 34:180–186PubMed
2.
go back to reference Kamal NM, Jouini R, Yahya S, Haiba M (2011) Benign intrascrotal lipoblastoma in a 4-month-old infant: a case report and review of literature. J Pediatr Surg 46:e9–e12PubMed Kamal NM, Jouini R, Yahya S, Haiba M (2011) Benign intrascrotal lipoblastoma in a 4-month-old infant: a case report and review of literature. J Pediatr Surg 46:e9–e12PubMed
3.
go back to reference Bancroft LW, Kransdorf MJ, Peterson JJ, O’Connor MI (2006) Benign fatty tumors: classification, clinical course, imaging appearance, and treatment. Skelet Radiol 35:719–733 Bancroft LW, Kransdorf MJ, Peterson JJ, O’Connor MI (2006) Benign fatty tumors: classification, clinical course, imaging appearance, and treatment. Skelet Radiol 35:719–733
4.
go back to reference Yoshida H, Miyachi M, Ouchi K, Kuwahara Y, Tsuchiya K, Iehara T et al (2014) Identification of COL3A1 and RAB2A as novel translocation partner genes of PLAG1 in lipoblastoma. Genes Chromosome Cancer 53:606–611 Yoshida H, Miyachi M, Ouchi K, Kuwahara Y, Tsuchiya K, Iehara T et al (2014) Identification of COL3A1 and RAB2A as novel translocation partner genes of PLAG1 in lipoblastoma. Genes Chromosome Cancer 53:606–611
5.
go back to reference Nitta Y, Miyachi M, Tomida A, Sugimoto Y, Nakagawa N, Yoshida H et al (2019) Identification of a novel BOC-PLAG1 fusion gene in a case of lipoblastoma. Biochem Biophys Res Commun 512:49–52PubMed Nitta Y, Miyachi M, Tomida A, Sugimoto Y, Nakagawa N, Yoshida H et al (2019) Identification of a novel BOC-PLAG1 fusion gene in a case of lipoblastoma. Biochem Biophys Res Commun 512:49–52PubMed
6.
go back to reference Gisselsson D, Hibbard MK, Dal Cin P, Sciot R, Hsi B-L, Kozakewich HP et al (2001) PLAG1 alterations in lipoblastoma: involvement in varied mesenchymal cell types and evidence for alternative oncogenic mechanisms. Am J Pathol 159:955–962PubMedPubMedCentral Gisselsson D, Hibbard MK, Dal Cin P, Sciot R, Hsi B-L, Kozakewich HP et al (2001) PLAG1 alterations in lipoblastoma: involvement in varied mesenchymal cell types and evidence for alternative oncogenic mechanisms. Am J Pathol 159:955–962PubMedPubMedCentral
7.
go back to reference Toma R, Ferris S, Coombs CJ, Johnstone BR, McCombe DB (2010) Lipoblastoma: an important differential diagnosis of tumours of the hand in children. J Plast Surg Hand Surg 44:257–259PubMed Toma R, Ferris S, Coombs CJ, Johnstone BR, McCombe DB (2010) Lipoblastoma: an important differential diagnosis of tumours of the hand in children. J Plast Surg Hand Surg 44:257–259PubMed
8.
go back to reference López-Ferrer P, Jiménez-Heffernan JA, Yébenes L, Vicandi B, Viguer JM (2005) Fine-needle aspiration cytology of lipoblastoma: a report of two cases. Diagn Cytopathol 32:32–34PubMed López-Ferrer P, Jiménez-Heffernan JA, Yébenes L, Vicandi B, Viguer JM (2005) Fine-needle aspiration cytology of lipoblastoma: a report of two cases. Diagn Cytopathol 32:32–34PubMed
9.
go back to reference Fallon SC, Brandt ML, Rodriguez JR, Vasudevan SA, Lopez ME, Hicks MJ et al (2013) Cytogenetic analysis in the diagnosis and management of lipoblastomas: results from a single institution. J Surg Res 184:341–346PubMed Fallon SC, Brandt ML, Rodriguez JR, Vasudevan SA, Lopez ME, Hicks MJ et al (2013) Cytogenetic analysis in the diagnosis and management of lipoblastomas: results from a single institution. J Surg Res 184:341–346PubMed
10.
go back to reference Kaneyama K, Yamataka A, Okazaki T, Lane GJ, Miyano T (2006) Magnetic resonance imaging in lipoblastoma: can it be a diagnostic modality? Asian J Surg 29:198–201PubMed Kaneyama K, Yamataka A, Okazaki T, Lane GJ, Miyano T (2006) Magnetic resonance imaging in lipoblastoma: can it be a diagnostic modality? Asian J Surg 29:198–201PubMed
11.
go back to reference Reiseter T, Nordshus T, Borthne A, Roald B, Naess P, Schistad O (1999) Lipoblastoma: MRI appearances of a rare paediatric soft tissue tumour. Pediatr Radiol 29:542–545PubMed Reiseter T, Nordshus T, Borthne A, Roald B, Naess P, Schistad O (1999) Lipoblastoma: MRI appearances of a rare paediatric soft tissue tumour. Pediatr Radiol 29:542–545PubMed
12.
go back to reference Ching A, Lee S, Chan Y (2002) Diagnosing paediatric mediastinal lipoblastoma using ultrasound-guided percutaneous needle biopsy: review and report. Clin Imaging 26:23–26PubMed Ching A, Lee S, Chan Y (2002) Diagnosing paediatric mediastinal lipoblastoma using ultrasound-guided percutaneous needle biopsy: review and report. Clin Imaging 26:23–26PubMed
13.
go back to reference Kirkham YA, Yarbrough CM, Salle JLP, Allen LM (2013) A rare case of inguinolabial lipoblastoma in a 13-month-old female. J Pediatr Urol 9:e64–e67PubMed Kirkham YA, Yarbrough CM, Salle JLP, Allen LM (2013) A rare case of inguinolabial lipoblastoma in a 13-month-old female. J Pediatr Urol 9:e64–e67PubMed
14.
go back to reference Mognato G, Cecchetto G, Carli M, Talenti E, d'Amore E, Pederzini F et al (2000) Is surgical treatment of lipoblastoma always necessary? J Pediatr Surg 35:1511–1513PubMed Mognato G, Cecchetto G, Carli M, Talenti E, d'Amore E, Pederzini F et al (2000) Is surgical treatment of lipoblastoma always necessary? J Pediatr Surg 35:1511–1513PubMed
15.
go back to reference Pham NS, Poirier B, Fuller SC, Dublin AB, Tollefson TT (2010) Pediatric lipoblastoma in the head and neck: a systematic review of 48 reported cases. Int J Pediatr Otorhinolaryngol 74:723–728PubMed Pham NS, Poirier B, Fuller SC, Dublin AB, Tollefson TT (2010) Pediatric lipoblastoma in the head and neck: a systematic review of 48 reported cases. Int J Pediatr Otorhinolaryngol 74:723–728PubMed
16.
go back to reference Moher D, Liberati A, Tetzlaff J, Altman DG (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 151:264–269PubMed Moher D, Liberati A, Tetzlaff J, Altman DG (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 151:264–269PubMed
17.
go back to reference Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J (2003) Methodological index for non-randomized studies (MINORS): development and validation of a new instrument. ANZ J Surg 73:712–716PubMed Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J (2003) Methodological index for non-randomized studies (MINORS): development and validation of a new instrument. ANZ J Surg 73:712–716PubMed
18.
go back to reference Hashimoto S, Kikuta K, Sekita T, Nakayama R, Takayama S, Sasaki A et al (2017) A giant popliteal lipoblastoma in a 23-month-old girl: a case report. J Med Case Rep 11:338PubMedPubMedCentral Hashimoto S, Kikuta K, Sekita T, Nakayama R, Takayama S, Sasaki A et al (2017) A giant popliteal lipoblastoma in a 23-month-old girl: a case report. J Med Case Rep 11:338PubMedPubMedCentral
19.
go back to reference DerSimonian R, Laird N (1986) Meta-analysis in clinical trials. Control Clin Trials 7:177–188PubMed DerSimonian R, Laird N (1986) Meta-analysis in clinical trials. Control Clin Trials 7:177–188PubMed
20.
go back to reference Cummings P (2009) The relative merits of risk ratios and odds ratios. Arch Pediatr Adolesc Med 163:438–445PubMed Cummings P (2009) The relative merits of risk ratios and odds ratios. Arch Pediatr Adolesc Med 163:438–445PubMed
21.
go back to reference Hozo SP, Djulbegovic B, Hozo I (2005) Estimating the mean and variance from the median, range, and the size of a sample. BMC Med Res Methodol 5:13PubMedPubMedCentral Hozo SP, Djulbegovic B, Hozo I (2005) Estimating the mean and variance from the median, range, and the size of a sample. BMC Med Res Methodol 5:13PubMedPubMedCentral
22.
go back to reference Hicks J, Dilley A, Patel D, Barrish J, Zhu SH, Brandt M (2001) Lipoblastoma and lipoblastomatosis in infancy and childhood: histopathologic, ultrastructural, and cytogenetic features. Ultrastruct Pathol 25:233–321 Hicks J, Dilley A, Patel D, Barrish J, Zhu SH, Brandt M (2001) Lipoblastoma and lipoblastomatosis in infancy and childhood: histopathologic, ultrastructural, and cytogenetic features. Ultrastruct Pathol 25:233–321
23.
go back to reference Kok KY, Telisinghe P (2010) Lipoblastoma: clinical features, treatment, and outcome. World J Surg 34:1517–1522PubMed Kok KY, Telisinghe P (2010) Lipoblastoma: clinical features, treatment, and outcome. World J Surg 34:1517–1522PubMed
24.
go back to reference de Saint Aubain Somerhausen N, Coindre JM, Debiec-Rychter M, Delplace J, Sciot R (2008) Lipoblastoma in adolescents and young adults: report of six cases with FISH analysis. Histopathology 52:294–298PubMed de Saint Aubain Somerhausen N, Coindre JM, Debiec-Rychter M, Delplace J, Sciot R (2008) Lipoblastoma in adolescents and young adults: report of six cases with FISH analysis. Histopathology 52:294–298PubMed
25.
go back to reference Abdul-Ghafar J, Ahmad Z, Tariq MU, Kayani N, Uddin N (2018) Lipoblastoma: a clinicopathologic review of 23 cases from a major tertiary care center plus detailed review of literature. BMC Res Notes 11:42PubMedPubMedCentral Abdul-Ghafar J, Ahmad Z, Tariq MU, Kayani N, Uddin N (2018) Lipoblastoma: a clinicopathologic review of 23 cases from a major tertiary care center plus detailed review of literature. BMC Res Notes 11:42PubMedPubMedCentral
26.
go back to reference Cappellesso R, d’Amore ES, Dall’Igna P, Guzzardo V, Vassarotto E, Rugge M et al (2016) Immunohistochemical expression of p16 in lipoblastomas. Hum Pathol 47:64–69PubMed Cappellesso R, d’Amore ES, Dall’Igna P, Guzzardo V, Vassarotto E, Rugge M et al (2016) Immunohistochemical expression of p16 in lipoblastomas. Hum Pathol 47:64–69PubMed
27.
go back to reference Chun YS, Kim WK, Park KW, Lee SC, Jung SE (2001) Lipoblastoma. J Pediatr Surg 36:905–907PubMed Chun YS, Kim WK, Park KW, Lee SC, Jung SE (2001) Lipoblastoma. J Pediatr Surg 36:905–907PubMed
28.
go back to reference Chung E, Enzinger F (1973) Benign lipoblastomatosis. An analysis of 35 cases. Cancer 32:482–492PubMed Chung E, Enzinger F (1973) Benign lipoblastomatosis. An analysis of 35 cases. Cancer 32:482–492PubMed
29.
go back to reference Coffin CM, Lowichik A, Putnam A (2009) Lipoblastoma (LPB): a clinicopathologic and immunohistochemical analysis of 59 cases. Am J Surg Pathol 33:1705–1712PubMed Coffin CM, Lowichik A, Putnam A (2009) Lipoblastoma (LPB): a clinicopathologic and immunohistochemical analysis of 59 cases. Am J Surg Pathol 33:1705–1712PubMed
30.
go back to reference Collins MH, Chatten J (1997) Lipoblastoma/lipoblastomatosis: a clinicopathologic study of 25 tumors. Am J Surg Pathol 21:1131–1137PubMed Collins MH, Chatten J (1997) Lipoblastoma/lipoblastomatosis: a clinicopathologic study of 25 tumors. Am J Surg Pathol 21:1131–1137PubMed
31.
go back to reference Dilley AV, Patel DL, Hicks MJ, Brandt ML (2001) Lipoblastoma: pathophysiology and surgical management. J Pediatr Surg 36:229–231PubMed Dilley AV, Patel DL, Hicks MJ, Brandt ML (2001) Lipoblastoma: pathophysiology and surgical management. J Pediatr Surg 36:229–231PubMed
32.
go back to reference Ferreira J, Esteves G, Fonseca R, Martins C, André S, Lemos MM (2017) Fine-needle aspiration of lipoblastoma: cytological, molecular, and clinical features. Cancer Cytopathol 125:934–939PubMed Ferreira J, Esteves G, Fonseca R, Martins C, André S, Lemos MM (2017) Fine-needle aspiration of lipoblastoma: cytological, molecular, and clinical features. Cancer Cytopathol 125:934–939PubMed
33.
go back to reference Han JW, Kim H, Youn JK, Oh C, Jung SE, Park KW et al (2017) Analysis of clinical features of lipoblastoma in children. Pediatr Hematol Oncol 34:212–220PubMed Han JW, Kim H, Youn JK, Oh C, Jung SE, Park KW et al (2017) Analysis of clinical features of lipoblastoma in children. Pediatr Hematol Oncol 34:212–220PubMed
34.
go back to reference Jung S-M, Chang P-Y, Luo C-C, Huang C-S, Lai J-Y, Hsueh C (2005) Lipoblastoma/lipoblastomatosis: a clinicopathologic study of 16 cases in Taiwan. Pediatr Surg Int 21:809–812PubMed Jung S-M, Chang P-Y, Luo C-C, Huang C-S, Lai J-Y, Hsueh C (2005) Lipoblastoma/lipoblastomatosis: a clinicopathologic study of 16 cases in Taiwan. Pediatr Surg Int 21:809–812PubMed
35.
go back to reference Kerkeni Y, Sahnoun L, Ksia A, Hidouri S, Chahed J, Krichen I et al (2014) Lipoblastoma in childhood: about 10 cases. Afr J Paediatr Surg 11:32–34PubMed Kerkeni Y, Sahnoun L, Ksia A, Hidouri S, Chahed J, Krichen I et al (2014) Lipoblastoma in childhood: about 10 cases. Afr J Paediatr Surg 11:32–34PubMed
36.
go back to reference McVay MR, Keller JE, Wagner CW, Jackson RJ, Smith SD (2006) Surgical management of lipoblastoma. J Pediatr Surg 41:1067–1071PubMed McVay MR, Keller JE, Wagner CW, Jackson RJ, Smith SD (2006) Surgical management of lipoblastoma. J Pediatr Surg 41:1067–1071PubMed
37.
go back to reference Mentzel T, Calonje E, Fletcher C (1993) Lipoblastoma and lipoblastomatosis: a clinicopathological study of 14 cases. Histopathology 23:527–533PubMed Mentzel T, Calonje E, Fletcher C (1993) Lipoblastoma and lipoblastomatosis: a clinicopathological study of 14 cases. Histopathology 23:527–533PubMed
38.
go back to reference Miller GG, Yanchar NL, Magee JF, Blair GK (1998) Lipoblastoma and liposarcoma in children: an analysis of 9 cases and a review of the literature. Can J Surg 41:455–458PubMedPubMedCentral Miller GG, Yanchar NL, Magee JF, Blair GK (1998) Lipoblastoma and liposarcoma in children: an analysis of 9 cases and a review of the literature. Can J Surg 41:455–458PubMedPubMedCentral
39.
go back to reference Mo Z, Xie X, Wang H, Qin H, Wei H, Li X (2017) Analysis of diagnosis and treatment of lipoblastomatosis. Sci China Life Sci 60:778–780PubMed Mo Z, Xie X, Wang H, Qin H, Wei H, Li X (2017) Analysis of diagnosis and treatment of lipoblastomatosis. Sci China Life Sci 60:778–780PubMed
40.
go back to reference Moholkar S, Sebire NJ, Roebuck DJ (2006) Radiological–pathological correlation in lipoblastoma and lipoblastomatosis. Pediatr Radiol 36:851–856PubMed Moholkar S, Sebire NJ, Roebuck DJ (2006) Radiological–pathological correlation in lipoblastoma and lipoblastomatosis. Pediatr Radiol 36:851–856PubMed
41.
go back to reference Séguier-Lipszyc E, Baazov A, Fichman S, Ash S, Freud E (2018) Current management of lipoblastoma. Eur J Pediatr 177:237–241PubMed Séguier-Lipszyc E, Baazov A, Fichman S, Ash S, Freud E (2018) Current management of lipoblastoma. Eur J Pediatr 177:237–241PubMed
42.
go back to reference Speer AL, Schofield DE, Wang KS, Shin CE, Stein JE, Shaul DB et al (2008) Contemporary management of lipoblastoma. J Pediatr Surg 43:1295–1300PubMed Speer AL, Schofield DE, Wang KS, Shin CE, Stein JE, Shaul DB et al (2008) Contemporary management of lipoblastoma. J Pediatr Surg 43:1295–1300PubMed
43.
go back to reference Susam-Sen H, Yalcin B, Kutluk T, Cahit Tanyel F, Haliloglu M, Orhan D et al (2017) Lipoblastoma in children: review of 12 cases. Pediatr Int 59:545–550PubMed Susam-Sen H, Yalcin B, Kutluk T, Cahit Tanyel F, Haliloglu M, Orhan D et al (2017) Lipoblastoma in children: review of 12 cases. Pediatr Int 59:545–550PubMed
44.
go back to reference Koo TK, Li MY (2016) A guideline of selecting and reporting intraclass correlation coefficients for reliability research. J Chiropr Med 15:155–163PubMedPubMedCentral Koo TK, Li MY (2016) A guideline of selecting and reporting intraclass correlation coefficients for reliability research. J Chiropr Med 15:155–163PubMedPubMedCentral
46.
go back to reference Shi Y, Yang K, Zhang M, Yi C, Yu J (2019) Lipoblastoma of the left kidney: a case report and review of literature. Ann Transl Med 7:150PubMedPubMedCentral Shi Y, Yang K, Zhang M, Yi C, Yu J (2019) Lipoblastoma of the left kidney: a case report and review of literature. Ann Transl Med 7:150PubMedPubMedCentral
47.
go back to reference Fanna M, Rougemont A-L, Arni D, Toso C, Anooshiravani-Dumont M, Wildhaber BE (2019) Giant intrahepatic lipoblastoma in a child. J Pediatr 210:235–236.e1PubMed Fanna M, Rougemont A-L, Arni D, Toso C, Anooshiravani-Dumont M, Wildhaber BE (2019) Giant intrahepatic lipoblastoma in a child. J Pediatr 210:235–236.e1PubMed
Metadata
Title
Follow-up outcomes of pediatric patients who underwent surgical resection for lipoblastomas or lipoblastomatosis: a single-institution experience with a systematic review and meta-analysis
Authors
Dyda Dao
Anna J. Najor
Philip Y. Sun
Forough Farrokhyar
Christopher R. Moir
Michael B. Ishitani
Publication date
01-03-2020
Publisher
Springer Berlin Heidelberg
Published in
Pediatric Surgery International / Issue 3/2020
Print ISSN: 0179-0358
Electronic ISSN: 1437-9813
DOI
https://doi.org/10.1007/s00383-019-04612-z

Other articles of this Issue 3/2020

Pediatric Surgery International 3/2020 Go to the issue