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Middle ear lipoma mimicking a congenital cholesteatoma: A case report and review of the literature
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
Middle ear lipoma mimicking a congenital cholesteatoma: A case report and review of the literature
T
Floyd Buena, Chia-Huei Chub,c, Akira Ishiyamaa,∗ a
Department of Head & Neck Surgery, UCLA David Geffen School of Medicine, Los Angeles, CA, USA Department of Otolaryngology- Head & Neck Surgery, Taipei Veterans General Hospital, Taiwan c Department of Otolaryngology, National Yang Ming University School of Medicine, Taiwan b
A R T I C LE I N FO
A B S T R A C T
Keywords: Lipoma Middle ear Temporal bone mass
Objective: To describe a case of middle ear lipoma, review the current literature, and discuss the surgical approach. Methods: Published case reports in the English literature of lipomas restricted to the middle ear were reviewed. The presentation, location, and management of the middle ear lipomas were analyzed. Results: Histological examination of the resected middle ear lesion was compatible with lipoma. Review of the literature suggests middle ear lipomas are rare and involve the epitympanum. Conclusion: Lipomas should be included in the differential diagnosis for middle ear lesions. Adequate surgical exposure can be achieved through a transcanal approach, with particular attention to carefully elevate the tympanic membrane off the malleus, preserving the integrity of the ossicular chain.
1. Introduction
otolaryngologist. Presence of a white lesion was detected in the anterior epitympanum on examination in the right ear. The patient had an audiogram using behavioral testing which demonstrated speech reception threshold at 25dB. CT scan of the temporal bone demonstrated a right middle ear soft tissue density involving the epitympanum and it was considered to be compatible with the diagnosis of congenital cholesteatoma, therefore, no further imaging studies were indicated (Fig. 1). This report is a review of a single patient's experience and management for middle ear lipoma at the UCLA Ronald Reagan Medical Center. As such, this study is exempted from the Institutional Review Board. All patient information has been de-identified.
One of the most common neoplasms of mesenchymal origin are lipomas [1]. Lipomas are benign tumors composed of mature adipocytes within lobules, enclosed by a fibrous capsule and located primarily in subcutaneous tissue. Lipomas tend to be slow growing and cause few symptoms. Thirteen percent of lipomas are found in the head and neck and a majority develop in the posterior neck region [1]. There have been instances of lipomas occurring within the temporal bone, specifically, in the internal auditory canal as well as the cerebellopontine angle [2–4]. Additionally, there are several case reports of lipomas occurring in the middle ear. Here, we report a case of a middle ear lipoma in a pediatric patient, describe our treatment approach for the mass and review the current literature regarding middle ear lipomas. 2. Materials and methods 2.1. Subject A seventeen-month-old female with a history of cleft palate was referred to our clinic for evaluation of probable congenital cholesteatoma. She initially presented with recurrent serous otitis media and underwent ventilation tube placement by an experienced pediatric ∗
2.2. Surgical approach Otomicroscopic examination of the right ear revealed a soft tissue lesion in the anterior epitypanum compatible with congenital cholesteatoma (Fig. 2A). After the tympanomeatal flap was elevated and the tympanic membrane was completely lifted off the malleus, the lesion grossly had the appearance of a lipoma (Fig. 2B). It was saddled in the anterior superior mesotympanum and involved the anterior epitympanum. We excised the lesion completely without disrupting the integrity of the middle ear ossicles.
Corresponding author. 10833 Le Conte Ave CHS 62-132, Los Angeles, CA, 90095, USA. E-mail address: [email protected] (A. Ishiyama).
https://doi.org/10.1016/j.ijporl.2018.09.021 Received 13 March 2018; Received in revised form 18 September 2018; Accepted 21 September 2018 Available online 22 September 2018 0165-5876/ © 2018 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
Fig. 1. CT temporal bone without contrast. Serial axial cuts starting superiorly (A) to inferiorly (B) demonstrating intact middle ear structures with an effusion and soft tissue density in the right middle ear. Coronal view starting more anteriorly (C) to posteriorly (D) demonstrating the soft tissue density in the anterior epitympanum and anterior superior mesotympanum. Mass is depicted by white arrow.
Fig. 2. Intraoperative images of the mass. (A) A yellowish mass (white arrow) is seen in the anterior mesotympanum without evidence of erosion of the tympanic membrane. (B) The tympanic membrane (blue arrow) was carefully dissected from the malleus (red arrow) revealing a well circumscribed lipomatous appearing mass. (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)
recurrence of the lipoma. The patient demonstrated speech detection in bilateral ears at 20dB. A total of seven reported cases of middle ear lipoma were reviewed (Table 1). Including the present report, six out of eight are pediatric cases. Four out of the seven case reports demonstrate a lipoma within the epitympanum. The anterior location of the lipoma in the present patient appears consistent with previous reports. All patients underwent surgical excision: five underwent transcanal approach while two underwent mastoidectomy. Additionally, removal of the incus and malleus were noted in two case reports during surgical excision requiring ossicular chain reconstruction.
2.3. Literature review A literature search was performed in Pubmed with key words that included “middle ear” and “lipoma”. Case reports that describe patients with histologically proven lipomas originating from the middle ear were included in the review. A total of seven case reports were found in the English literature between the years 1985 and 2017. The presentation of each patient, location of the middle ear lipoma, and surgical technique were analyzed. 3. Results
4. Discussion
During surgical excision of the soft tissue mass in the present case, there was no evidence of ossicular chain erosion. The described surgical approach was sufficient for complete excision, without need to remove the malleus or incus for greater exposure, or for increased access to the mass. Histological analysis demonstrated mature adipocytes consistent with a lipoma (Fig. 3). Five months later, the patient had a well healed tympanic membrane without evidence of perforation, effusion or
To our knowledge, there have only been seven reported cases of middle ear lipoma in the literature. The present report represents a case of a young patient with a middle ear lipoma, suspected to be a congenital cholesteatoma. All reported cases of middle ear lipoma demonstrated hearing loss, most commonly conductive hearing loss. 111
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
inflammation of the facial nerve. This is supported by the fact that the patient did have interval improvement with medical management prior to surgical resection of the lipoma. Interestingly, Luetje et al. discovered that 17% of 240 cadaver temporal bones showed fat deposition adjacent to the vertical segment of the facial nerve in the mastoid [7]. It is possible that such fat deposition may also be a source of lipoma development. During development, the ectoderm of the first branchial groove proliferates medially which eventually contributes to the formation of the external auditory canal [8–10]. Meanwhile, the endoderm of the first pharyngeal pouch extends laterally as the tubotympanic recess. The most proximal part of the recess forms the eustachian tube and the most distal end forms the middle ear. The interface between the medial most ectoderm and the lateral most endoderm forms the tympanic membrane. The ossicles are derived from the neural crest mesenchyme from the first and second branchial arch. In an analysis of temporal bones from fetuses and children, Tono et al. describe the independent development of the epitympanum which forms after birth as a cavity filled with mesenchyme [11]. The mesenchyme may persist years after birth and is slowly resorbed with pneumatization of the epitympanum. Interestingly, these authors comment that several epitympanum samples were filled with fibrous connective tissue accompanied by small nests of adipocytes. This suggests that retained embryonic mesenchymal tissue may differentiate to adipose tissue, leading to the development of a middle ear lipoma. This appears consistent with previous case reports of middle ear lipomas involving the epitympanum. Because there are such few cases, lipoma is not commonly in the initial differential diagnosis of a soft tissue mass in the middle ear. In our workup, the patient's clinical history, exam and CT findings were strongly in favor of congenital cholesteatoma. While an MRI is not routinely performed for cholesteatoma, T1 sequence on MRI may have been able to differentiate a lipoma and cholesteatoma. Finally, there is no standard treatment approach for these rare middle ear lipomas. In the present report, the lipoma was located in the mesotympanum involving the anterior epitympanum. To achieve complete removal of the lipoma, two of the seven case reports in the literature describe removal of the malleus and incus, both of which were intact, to gain access to the epitympanum. In contrast, our surgical approach entailed careful release of the tympanic membrane from the malleus handle. This facilitated access to the mass without compromising the integrity of the native ossicular chain and circumvented the
Fig. 3. H&E staining of mass, which demonstrates mature adipose tissue surrounded by fibrovascular tissue consistent with lipoma. No cholesteatoma is identified.
While several case reports initially suspected cholesteatoma due to a soft tissue density in the middle ear, other cases had unusual presentations. Stegehuis et al. describes a 64-year-old female who underwent a stapedectomy 18 years ago with adequate hearing improvement, though later developed progressive conductive hearing loss [5]. She initially presented with shortness of breath and dysphagia. She was found to have a polyp originating from the middle ear extending through the eustachian tube and into the pharynx. The final pathology of the polyp demonstrated lipoma. As polyps that occur after stapedectomy tend to be granulomatous and not lipomatous, the authors concluded that the lipoma and history of stapedectomy were coincidental. Kasbekar et al. presented a case of a 33-year-old male who had a history of mastoidectomy for a cholesteatoma as a child who, as an adult, presented with hemifacial weakness and acute otitis media [6]. A middle ear exploration revealed a lipoma adjacent to the horizontal segment of the facial nerve. The authors believe the lipoma may have obstructed the eustachian tube, resulting in acute otitis media and local
Table 1 Compilation of published case reports of middle ear lipoma including the present report. Age
Presenting Symptom
Location of Lipoma and Findings
Reference
64 year old female
Antero-superior medial wall of middle ear with extension into eustachian tube causing intermittent airway obstruction. Surgical approach unclear.
Stegehuis HR et al. [5]
Middle ear exploration showed anterior epitympanum mass, filing entire middle ear space. Ossicles intact.
Selesnick SH et al. [12]
Middle ear exploration showed high anterior epitympanum mass with occlusion of eustachian tube. Tympanomastoidectomy showed anterior medial mass, obstructing eustachian tube without involvement of ossicles. Mastoidectomy showed anterior middle ear mass adjacent to horizontal portion of the facial nerve.
Abdullah V et al. [13] Edmonds JL et al. [14] Kasbekar AV et al. [6]
11 month old male
Shortness of breath and dysphagia secondary to lipoma extending from eustachian tube into the pharynx. Patient had longstanding conductive hearing loss. History of atonic diplegia, motor, developmental and speech delay. Patient had bilateral mild to moderate conductive hearing loss. Speech and language delay, bilateral conductive hearing loss, refractory to pressure equalizing tubes. Craniofacial abnormalities and developmental delay, eustachian tube dysfunction with multiple tube placement. History of cholesteatoma and modified radical mastoidectomy as a child. Later developed right sided hemifacial paresis, otitis media and moderate conductive hearing loss. Down syndrome, failed new born hearing screening at 2 months and found to have bilateral effusion.
Karas A et al. [15]
6 year old female
Down syndrome, speech and language delay with moderate conductive loss.
17 month old female
History of cleft palate, initially though to have bilateral congenital cholesteatoma.
Middle ear exploration showed anterior superior quadrant mass. Excision of incus and malleus head for access to epitympanum, OCR. Note, patient had bilateral lipomas. Middle ear exploration showed anterior middle ear mass, ossicles intact. Incus and malleus head removed for access to epitympanum and OCR performed. Middle ear exploration showed anterior superior mesotympanum mass with involvement of the epitympanum.
4 year old female 5 year old female 7 year old female 33 year old male
112
Aldosari et al. [16] Present report.
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
need to perform reconstruction with a prosthetic device. If indeed the majority of middle ear lipomas originate in the epitympanum and are discovered early, the same surgical approach can be applied.
[3] A. Bacciu, F. Di Lella, E. Ventura, E. Pasanisi, A. Russo, M. Sanna, Lipomas of the internal auditory canal and cerebellopontine angle, Ann. Otol. Rhinol. Laryngol. 123 (2014) 58–64. [4] C.L. Truwit, A.J. Barkovich, Pathogenesis of intracranial lipoma: an MR study in 42 patients, AJNR Am J Neuroradiol 11 (1990) 665–674. [5] H.R. Stegehuis, A.M. Guy, K.R. Anderson, Middle-ear lipoma presenting as airways obstruction: case report and review of literature, J. Laryngol. Otol. 99 (1985) 589–591. [6] A.V. Kasbekar, N. Donnelly, P. Axon, Facial nerve palsy secondary to middle-ear lipoma, J. Laryngol. Otol. 122 (2008) e14. [7] C.M. Luetje, M.M. Milbrath, F.H. Linthicum Jr., Juxtaposition lipoma to the mastoid seventh nerve, Laryngoscope 104 (1994) 135–139. [8] J.T. Johnson, C.A. Rosen, B.J. Bailey, Bailey's Head and Neck Surgery–otolaryngology vol. 2, Wolters Kluwer Health/Lippincott Williams & Wilkins, Philadelphia, Pa, 2014, p. 1 online resource. [9] J.J. Ballenger, J.B. Snow, Ballenger's Otorhinolaryngology: Head and Neck Surgery, BC Decker, Hamilton, Ontario, 2003, p. 1616 xv. [10] M. Mallo, Embryological and genetic aspects of middle ear development, Int. J. Dev. Biol. 42 (1998) 11–22. [11] T. Tono, P.A. Schachern, T. Morizono, M.M. Paparella, T. Morimitsu, Developmental anatomy of the supratubal recess in temporal bones from fetuses and children, Am. J. Otol. 17 (1996) 99–107. [12] S.H. Selesnick, D.R. Edelstein, S.C. Parisier, Lipoma of the middle ear: an unusual presentation in a 4-year-old child, Otolaryngol. Head Neck Surg. 102 (1990) 82–84. [13] V. Abdullah, P. Williamson, A. Gallimore, N.S. Shah, Middle ear lipoma, J. Laryngol. Otol. 107 (1993) 1151–1152. [14] J.L. Edmonds, J.M. Woodroof, G.A. Ator, Middle-ear lipoma as a cause of otomastoiditis, J. Laryngol. Otol. 111 (1997) 1162–1165. [15] A. Karas, WD, B. Rabin, E. Cham, N.M. Young, An unusual pediatric case of bilateral lipoma of the attic and middle ear, Int. J. Pediatr. Otorhinolaryngol. Extra 6 (2011) 206–208. [16] B. Aldosari, Lipoma of the middle ear: an unusual presentation in a 6 year old child, Acta Otorrinolaringol. Esp. 65 (2014) 381–382.
5. Conclusion Lipomas are common to the head and neck region but are exceedingly rare in the middle ear. Middle ear lipomas may originate from retained mesenchymal tissue within the epitympanum. Complete excision can be performed through a transcanal approach with preservation of the ossicular chain. Financial support None. Conflicts of interest None. References [1] M.H. El-Monem, A.H. Gaafar, E.A. Magdy, Lipomas of the head and neck: presentation variability and diagnostic work-up, J. Laryngol. Otol. 120 (2006) 47–55. [2] J.R. White, M.L. Carlson, J.J. Van Gompel, B.A. Neff, C.L. Driscoll, et al., Lipomas of the cerebellopontine angle and internal auditory canal: primum non nocere, Laryngoscope 123 (2013) 1531–1536.
113
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
Middle ear lipoma mimicking a congenital cholesteatoma: A case report and review of the literature
T
Floyd Buena, Chia-Huei Chub,c, Akira Ishiyamaa,∗ a
Department of Head & Neck Surgery, UCLA David Geffen School of Medicine, Los Angeles, CA, USA Department of Otolaryngology- Head & Neck Surgery, Taipei Veterans General Hospital, Taiwan c Department of Otolaryngology, National Yang Ming University School of Medicine, Taiwan b
A R T I C LE I N FO
A B S T R A C T
Keywords: Lipoma Middle ear Temporal bone mass
Objective: To describe a case of middle ear lipoma, review the current literature, and discuss the surgical approach. Methods: Published case reports in the English literature of lipomas restricted to the middle ear were reviewed. The presentation, location, and management of the middle ear lipomas were analyzed. Results: Histological examination of the resected middle ear lesion was compatible with lipoma. Review of the literature suggests middle ear lipomas are rare and involve the epitympanum. Conclusion: Lipomas should be included in the differential diagnosis for middle ear lesions. Adequate surgical exposure can be achieved through a transcanal approach, with particular attention to carefully elevate the tympanic membrane off the malleus, preserving the integrity of the ossicular chain.
1. Introduction
otolaryngologist. Presence of a white lesion was detected in the anterior epitympanum on examination in the right ear. The patient had an audiogram using behavioral testing which demonstrated speech reception threshold at 25dB. CT scan of the temporal bone demonstrated a right middle ear soft tissue density involving the epitympanum and it was considered to be compatible with the diagnosis of congenital cholesteatoma, therefore, no further imaging studies were indicated (Fig. 1). This report is a review of a single patient's experience and management for middle ear lipoma at the UCLA Ronald Reagan Medical Center. As such, this study is exempted from the Institutional Review Board. All patient information has been de-identified.
One of the most common neoplasms of mesenchymal origin are lipomas [1]. Lipomas are benign tumors composed of mature adipocytes within lobules, enclosed by a fibrous capsule and located primarily in subcutaneous tissue. Lipomas tend to be slow growing and cause few symptoms. Thirteen percent of lipomas are found in the head and neck and a majority develop in the posterior neck region [1]. There have been instances of lipomas occurring within the temporal bone, specifically, in the internal auditory canal as well as the cerebellopontine angle [2–4]. Additionally, there are several case reports of lipomas occurring in the middle ear. Here, we report a case of a middle ear lipoma in a pediatric patient, describe our treatment approach for the mass and review the current literature regarding middle ear lipomas. 2. Materials and methods 2.1. Subject A seventeen-month-old female with a history of cleft palate was referred to our clinic for evaluation of probable congenital cholesteatoma. She initially presented with recurrent serous otitis media and underwent ventilation tube placement by an experienced pediatric ∗
2.2. Surgical approach Otomicroscopic examination of the right ear revealed a soft tissue lesion in the anterior epitypanum compatible with congenital cholesteatoma (Fig. 2A). After the tympanomeatal flap was elevated and the tympanic membrane was completely lifted off the malleus, the lesion grossly had the appearance of a lipoma (Fig. 2B). It was saddled in the anterior superior mesotympanum and involved the anterior epitympanum. We excised the lesion completely without disrupting the integrity of the middle ear ossicles.
Corresponding author. 10833 Le Conte Ave CHS 62-132, Los Angeles, CA, 90095, USA. E-mail address: [email protected] (A. Ishiyama).
https://doi.org/10.1016/j.ijporl.2018.09.021 Received 13 March 2018; Received in revised form 18 September 2018; Accepted 21 September 2018 Available online 22 September 2018 0165-5876/ © 2018 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
Fig. 1. CT temporal bone without contrast. Serial axial cuts starting superiorly (A) to inferiorly (B) demonstrating intact middle ear structures with an effusion and soft tissue density in the right middle ear. Coronal view starting more anteriorly (C) to posteriorly (D) demonstrating the soft tissue density in the anterior epitympanum and anterior superior mesotympanum. Mass is depicted by white arrow.
Fig. 2. Intraoperative images of the mass. (A) A yellowish mass (white arrow) is seen in the anterior mesotympanum without evidence of erosion of the tympanic membrane. (B) The tympanic membrane (blue arrow) was carefully dissected from the malleus (red arrow) revealing a well circumscribed lipomatous appearing mass. (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)
recurrence of the lipoma. The patient demonstrated speech detection in bilateral ears at 20dB. A total of seven reported cases of middle ear lipoma were reviewed (Table 1). Including the present report, six out of eight are pediatric cases. Four out of the seven case reports demonstrate a lipoma within the epitympanum. The anterior location of the lipoma in the present patient appears consistent with previous reports. All patients underwent surgical excision: five underwent transcanal approach while two underwent mastoidectomy. Additionally, removal of the incus and malleus were noted in two case reports during surgical excision requiring ossicular chain reconstruction.
2.3. Literature review A literature search was performed in Pubmed with key words that included “middle ear” and “lipoma”. Case reports that describe patients with histologically proven lipomas originating from the middle ear were included in the review. A total of seven case reports were found in the English literature between the years 1985 and 2017. The presentation of each patient, location of the middle ear lipoma, and surgical technique were analyzed. 3. Results
4. Discussion
During surgical excision of the soft tissue mass in the present case, there was no evidence of ossicular chain erosion. The described surgical approach was sufficient for complete excision, without need to remove the malleus or incus for greater exposure, or for increased access to the mass. Histological analysis demonstrated mature adipocytes consistent with a lipoma (Fig. 3). Five months later, the patient had a well healed tympanic membrane without evidence of perforation, effusion or
To our knowledge, there have only been seven reported cases of middle ear lipoma in the literature. The present report represents a case of a young patient with a middle ear lipoma, suspected to be a congenital cholesteatoma. All reported cases of middle ear lipoma demonstrated hearing loss, most commonly conductive hearing loss. 111
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
inflammation of the facial nerve. This is supported by the fact that the patient did have interval improvement with medical management prior to surgical resection of the lipoma. Interestingly, Luetje et al. discovered that 17% of 240 cadaver temporal bones showed fat deposition adjacent to the vertical segment of the facial nerve in the mastoid [7]. It is possible that such fat deposition may also be a source of lipoma development. During development, the ectoderm of the first branchial groove proliferates medially which eventually contributes to the formation of the external auditory canal [8–10]. Meanwhile, the endoderm of the first pharyngeal pouch extends laterally as the tubotympanic recess. The most proximal part of the recess forms the eustachian tube and the most distal end forms the middle ear. The interface between the medial most ectoderm and the lateral most endoderm forms the tympanic membrane. The ossicles are derived from the neural crest mesenchyme from the first and second branchial arch. In an analysis of temporal bones from fetuses and children, Tono et al. describe the independent development of the epitympanum which forms after birth as a cavity filled with mesenchyme [11]. The mesenchyme may persist years after birth and is slowly resorbed with pneumatization of the epitympanum. Interestingly, these authors comment that several epitympanum samples were filled with fibrous connective tissue accompanied by small nests of adipocytes. This suggests that retained embryonic mesenchymal tissue may differentiate to adipose tissue, leading to the development of a middle ear lipoma. This appears consistent with previous case reports of middle ear lipomas involving the epitympanum. Because there are such few cases, lipoma is not commonly in the initial differential diagnosis of a soft tissue mass in the middle ear. In our workup, the patient's clinical history, exam and CT findings were strongly in favor of congenital cholesteatoma. While an MRI is not routinely performed for cholesteatoma, T1 sequence on MRI may have been able to differentiate a lipoma and cholesteatoma. Finally, there is no standard treatment approach for these rare middle ear lipomas. In the present report, the lipoma was located in the mesotympanum involving the anterior epitympanum. To achieve complete removal of the lipoma, two of the seven case reports in the literature describe removal of the malleus and incus, both of which were intact, to gain access to the epitympanum. In contrast, our surgical approach entailed careful release of the tympanic membrane from the malleus handle. This facilitated access to the mass without compromising the integrity of the native ossicular chain and circumvented the
Fig. 3. H&E staining of mass, which demonstrates mature adipose tissue surrounded by fibrovascular tissue consistent with lipoma. No cholesteatoma is identified.
While several case reports initially suspected cholesteatoma due to a soft tissue density in the middle ear, other cases had unusual presentations. Stegehuis et al. describes a 64-year-old female who underwent a stapedectomy 18 years ago with adequate hearing improvement, though later developed progressive conductive hearing loss [5]. She initially presented with shortness of breath and dysphagia. She was found to have a polyp originating from the middle ear extending through the eustachian tube and into the pharynx. The final pathology of the polyp demonstrated lipoma. As polyps that occur after stapedectomy tend to be granulomatous and not lipomatous, the authors concluded that the lipoma and history of stapedectomy were coincidental. Kasbekar et al. presented a case of a 33-year-old male who had a history of mastoidectomy for a cholesteatoma as a child who, as an adult, presented with hemifacial weakness and acute otitis media [6]. A middle ear exploration revealed a lipoma adjacent to the horizontal segment of the facial nerve. The authors believe the lipoma may have obstructed the eustachian tube, resulting in acute otitis media and local
Table 1 Compilation of published case reports of middle ear lipoma including the present report. Age
Presenting Symptom
Location of Lipoma and Findings
Reference
64 year old female
Antero-superior medial wall of middle ear with extension into eustachian tube causing intermittent airway obstruction. Surgical approach unclear.
Stegehuis HR et al. [5]
Middle ear exploration showed anterior epitympanum mass, filing entire middle ear space. Ossicles intact.
Selesnick SH et al. [12]
Middle ear exploration showed high anterior epitympanum mass with occlusion of eustachian tube. Tympanomastoidectomy showed anterior medial mass, obstructing eustachian tube without involvement of ossicles. Mastoidectomy showed anterior middle ear mass adjacent to horizontal portion of the facial nerve.
Abdullah V et al. [13] Edmonds JL et al. [14] Kasbekar AV et al. [6]
11 month old male
Shortness of breath and dysphagia secondary to lipoma extending from eustachian tube into the pharynx. Patient had longstanding conductive hearing loss. History of atonic diplegia, motor, developmental and speech delay. Patient had bilateral mild to moderate conductive hearing loss. Speech and language delay, bilateral conductive hearing loss, refractory to pressure equalizing tubes. Craniofacial abnormalities and developmental delay, eustachian tube dysfunction with multiple tube placement. History of cholesteatoma and modified radical mastoidectomy as a child. Later developed right sided hemifacial paresis, otitis media and moderate conductive hearing loss. Down syndrome, failed new born hearing screening at 2 months and found to have bilateral effusion.
Karas A et al. [15]
6 year old female
Down syndrome, speech and language delay with moderate conductive loss.
17 month old female
History of cleft palate, initially though to have bilateral congenital cholesteatoma.
Middle ear exploration showed anterior superior quadrant mass. Excision of incus and malleus head for access to epitympanum, OCR. Note, patient had bilateral lipomas. Middle ear exploration showed anterior middle ear mass, ossicles intact. Incus and malleus head removed for access to epitympanum and OCR performed. Middle ear exploration showed anterior superior mesotympanum mass with involvement of the epitympanum.
4 year old female 5 year old female 7 year old female 33 year old male
112
Aldosari et al. [16] Present report.
International Journal of Pediatric Otorhinolaryngology 115 (2018) 110–113
F. Buen et al.
need to perform reconstruction with a prosthetic device. If indeed the majority of middle ear lipomas originate in the epitympanum and are discovered early, the same surgical approach can be applied.
[3] A. Bacciu, F. Di Lella, E. Ventura, E. Pasanisi, A. Russo, M. Sanna, Lipomas of the internal auditory canal and cerebellopontine angle, Ann. Otol. Rhinol. Laryngol. 123 (2014) 58–64. [4] C.L. Truwit, A.J. Barkovich, Pathogenesis of intracranial lipoma: an MR study in 42 patients, AJNR Am J Neuroradiol 11 (1990) 665–674. [5] H.R. Stegehuis, A.M. Guy, K.R. Anderson, Middle-ear lipoma presenting as airways obstruction: case report and review of literature, J. Laryngol. Otol. 99 (1985) 589–591. [6] A.V. Kasbekar, N. Donnelly, P. Axon, Facial nerve palsy secondary to middle-ear lipoma, J. Laryngol. Otol. 122 (2008) e14. [7] C.M. Luetje, M.M. Milbrath, F.H. Linthicum Jr., Juxtaposition lipoma to the mastoid seventh nerve, Laryngoscope 104 (1994) 135–139. [8] J.T. Johnson, C.A. Rosen, B.J. Bailey, Bailey's Head and Neck Surgery–otolaryngology vol. 2, Wolters Kluwer Health/Lippincott Williams & Wilkins, Philadelphia, Pa, 2014, p. 1 online resource. [9] J.J. Ballenger, J.B. Snow, Ballenger's Otorhinolaryngology: Head and Neck Surgery, BC Decker, Hamilton, Ontario, 2003, p. 1616 xv. [10] M. Mallo, Embryological and genetic aspects of middle ear development, Int. J. Dev. Biol. 42 (1998) 11–22. [11] T. Tono, P.A. Schachern, T. Morizono, M.M. Paparella, T. Morimitsu, Developmental anatomy of the supratubal recess in temporal bones from fetuses and children, Am. J. Otol. 17 (1996) 99–107. [12] S.H. Selesnick, D.R. Edelstein, S.C. Parisier, Lipoma of the middle ear: an unusual presentation in a 4-year-old child, Otolaryngol. Head Neck Surg. 102 (1990) 82–84. [13] V. Abdullah, P. Williamson, A. Gallimore, N.S. Shah, Middle ear lipoma, J. Laryngol. Otol. 107 (1993) 1151–1152. [14] J.L. Edmonds, J.M. Woodroof, G.A. Ator, Middle-ear lipoma as a cause of otomastoiditis, J. Laryngol. Otol. 111 (1997) 1162–1165. [15] A. Karas, WD, B. Rabin, E. Cham, N.M. Young, An unusual pediatric case of bilateral lipoma of the attic and middle ear, Int. J. Pediatr. Otorhinolaryngol. Extra 6 (2011) 206–208. [16] B. Aldosari, Lipoma of the middle ear: an unusual presentation in a 6 year old child, Acta Otorrinolaringol. Esp. 65 (2014) 381–382.
5. Conclusion Lipomas are common to the head and neck region but are exceedingly rare in the middle ear. Middle ear lipomas may originate from retained mesenchymal tissue within the epitympanum. Complete excision can be performed through a transcanal approach with preservation of the ossicular chain. Financial support None. Conflicts of interest None. References [1] M.H. El-Monem, A.H. Gaafar, E.A. Magdy, Lipomas of the head and neck: presentation variability and diagnostic work-up, J. Laryngol. Otol. 120 (2006) 47–55. [2] J.R. White, M.L. Carlson, J.J. Van Gompel, B.A. Neff, C.L. Driscoll, et al., Lipomas of the cerebellopontine angle and internal auditory canal: primum non nocere, Laryngoscope 123 (2013) 1531–1536.
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