primary hyperparathyroidism presenting as a brown tumor of...

1
58ESPE Poster presented at: P3-310 Jaida Manzoor DOI: 10.3252/pso.eu.58ESPE.2019 Bone, growth plate and mineral metabolism Primary Hyperparathyroidism Presenting as a Brown Tumor of Mandible in an Adolescent Girl - An Unusual presentation with Challenges and Outcome Jaida Manzoor, Saeed Ahmed, Nabila Talat, AbidAli Qureshi, Aisha Tahir Department of Endocrinology and Diabetes, Oral and Maxillofacial, Surgery The Children’s Hospital & Institute of Child Health Lahore University of Health Sciences Lahore Pakistan INTRODUCTION Brown tumor is a rare non-neoplastic focal giant cell lesion resulting due to increase osteolytic activity by excess of parathormone in cortical bone which is replaced by fibrovascular tissue, giant cells with hemorrhages and hemosiderin giving the brown color. It is a late stage bone sequelae of long- standing hyperparathyroidism (HPT) resulting in rare metabolic bone disease, Osteitis fibrosa cystica. HPT may occur in primary, secondary and tertiary forms. Parathyroid adenomas are the commonest cause in about 85% cases of Primary HPT. CASE PRESENTATION We present a referred case of 15-year-old girl with highly aggressive mandibular mass 4.0x3.0cm size creating swallowing difficulties, oral bleeding episodes, and anterior mandibular erosion with dislodgement of nine teeth, anterior canines, incisors, and premolars. The thorough clinical, biochemical, histopathology and comprehensive radiological assessment reveal left upper parathyroid adenoma causing a hyperparathyroid state and its long-standing existence led to “Brown tumor” of mandible. Serum Parathormone 516.00 pg/ml (N:11-67) Serum Calcium 12.5 mg/dl (N:8.4-10.2) Serum Phosphorus 2.39 mg/dl (N:3.0- 5.6) Alkaline Phosphatase 239 u/l 99m Technetium-MIBI Dual phase Parathyroid scan showed left upper parathyroid adenoma MRI craniofacial region showed non-infiltrative expansile soft tissue mass with anterior mandibular bone loss, teeth dislodgement and no other craniofacial involvement seen. Histopathology of specimen revealed predominant rich giant cell lesions with hemosiderin laden macrophages, fibroblastic stroma. Orthopantomogram(OPG): large unilocular radiolucency extending from mandibular right first premolar to left second premolar teeth. STEPS OF MANAGEMENT 1. The main stay of treatment was removal of left upper parathyroid adenoma and resulting in reversal of metabolic de-arrangements but subsequent follow-up showed incomplete regression of tumor. 2. Complete surgical excision of large disfiguring mandible residual and symptomatic mass was done successfully with peripheral osteotomy and placement of Kirschner (K) wire. 3. No evidence of recurrence was observed in one-year follow-up. Thereafter reconstructive surgery of anterior mandibular bone was performed using alloplastic devices and bone grafts from cadaveric source and synthetic bone fragments. 4. Alloplastic surgical membrane covered the whole augmented bone. 5. After few months of optimized healing, restoration of incisors, canines, premolar teeth was done initially by placing artificial denture followed by fixed dental prosthesis. OPG Before Intervention OPG after Intervention Use of Alloplastic Membrane Healed Augmented Anterior Mandible – Before Denture Application Successful Fixed Denture Placement OUTCOME REFERENCES q Goyal A, Boro H, Khadgawat R. Brown Tumor as an Index Presentation of. Severe Vitamin D Deficiency in a Teenage Girl. Cureus. 2018;10(5):e2722. q Zou H, Song L, Jia M, Wang L, Sun Y. Brown tumor of multiple facial bones associated with primary hyperparathyroidism. Medicine. 2018;97(33):e11877. q Swaminathan C. Surgical management of hyperparathyroid jaw tumour syndrome: A case report. International Journal of Applied Dental Sciences. 2018; 4(1):219-224. q Abdelrahman T. Brown Jaw Tumors: challenges and outcomes. International Surgery Journal. 2017;4(11):3586. q Kumar P, Jones K. Hyperparathyroid jaw tumour syndrome. Endocrine Abstracts. 2013;4(1):219- 224. CONCLUSION Such complications are rarely seen in presence of good medical standards and provision of advanced analytic facilities but still cases are encountered in underdeveloped countries with poor health facilities. The desirable aesthetic outcomes can only be produced with great expertise and can be achieved with biomaterial implants to replace, reconstruct and/or augment the tissue. Category: Bone, growth plate and mineral metabolism CORRESPONDING AUTHOR: Dr. Jaida Manzoor Email address: [email protected] The Children's Hospital & Institute of Child Health, Lahore, Pakistan. The Children’s Hospital & The Institute of Child Health Lahore

Upload: others

Post on 08-Jul-2020

6 views

Category:

Documents


0 download

TRANSCRIPT

Page 1: Primary Hyperparathyroidism Presenting as a Brown Tumor of ...abstracts.eurospe.org/hrp/0092/eposters/hrp0092p3-310_eposter.pdf · standing hyperparathyroidism (HPT) resulting in

58ES

PE

Poster presented at:P3-310

Jaida Manzoor DOI: 10.3252/pso.eu.58ESPE.2019

Bone, growth plate and mineral metabolism

Primary Hyperparathyroidism Presenting as a Brown Tumor ofMandible in an Adolescent Girl - An Unusual presentation withChallenges and OutcomeJaida Manzoor, Saeed Ahmed, Nabila Talat, Abid Ali Qureshi, Aisha Tahir

Department of Endocrinology and Diabetes, Oral and Maxillofacial, Surgery The Children’s Hospital & Institute of Child Health LahoreUniversity of Health Sciences Lahore Pakistan

INTRODUCTIONBrown tumor is a rare non-neoplastic focalgiant cell lesion resulting due to increaseosteolytic activity by excess ofparathormone in cortical bone which isreplaced by fibrovascular tissue, giant cellswith hemorrhages and hemosiderin givingthe brown color.

It is a late stage bone sequelae of long-standing hyperparathyroidism (HPT)resulting in rare metabolic bone disease,Osteitis fibrosa cystica.

HPT may occur in primary, secondary andtertiary forms. Parathyroid adenomas arethe commonest cause in about 85% casesof Primary HPT.

CASE PRESENTATION We present a referred case of 15-year-old girl with highly aggressivemandibular mass 4.0x3.0cm size creating swallowing difficulties, oralbleeding episodes, and anterior mandibular erosion with dislodgement ofnine teeth, anterior canines, incisors, and premolars.

The thorough clinical, biochemical, histopathology and comprehensiveradiological assessment reveal left upper parathyroid adenoma causing ahyperparathyroid state and its long-standing existence led to “Browntumor” of mandible.Serum Parathormone 516.00 pg/ml (N:11-67)Serum Calcium 12.5 mg/dl (N:8.4-10.2)Serum Phosphorus 2.39 mg/dl (N:3.0- 5.6)Alkaline Phosphatase 239 u/l

99mTechnetium-MIBI Dual phase Parathyroid scan showed left upperparathyroid adenomaMRI craniofacial region showed non-infiltrative expansile soft tissuemass with anterior mandibular bone loss, teeth dislodgement and noother craniofacial involvement seen.Histopathology of specimen revealed predominant rich giant cell lesionswith hemosiderin laden macrophages, fibroblastic stroma.

Orthopantomogram(OPG): large unilocularradiolucency extending from mandibular rightfirst premolar to left second premolar teeth.

STEPS OF MANAGEMENT

1. The main stay of treatment was removal ofleft upper parathyroid adenoma and resulting inreversal of metabolic de-arrangements butsubsequent follow-up showed incompleteregression of tumor.

2. Complete surgical excision of largedisfiguring mandible residual and symptomaticmass was done successfully with peripheralosteotomy and placement of Kirschner (K) wire.

3. No evidence of recurrence was observed inone-year follow-up. Thereafter reconstructivesurgery of anterior mandibular bone wasperformed using alloplastic devices and bonegrafts from cadaveric source and synthetic bonefragments.

4. Alloplastic surgical membrane covered thewhole augmented bone.

5. After few months of optimized healing,restoration of incisors, canines, premolar teethwas done initially by placing artificial denturefollowed by fixed dental prosthesis.

OPG Before Intervention

OPG after Intervention

Use of Alloplastic Membrane

Healed Augmented Anterior Mandible – Before Denture

Application

Successful Fixed Denture Placement

OU

TC

OM

E

REFERENCESq Goyal A, Boro H, Khadgawat R. Brown Tumor as an Index Presentation of. Severe Vitamin D

Deficiency in a Teenage Girl. Cureus. 2018;10(5):e2722.q Zou H, Song L, Jia M, Wang L, Sun Y. Brown tumor of multiple facial bones associated with

primary hyperparathyroidism. Medicine. 2018;97(33):e11877.q Swaminathan C. Surgical management of hyperparathyroid jaw tumour syndrome: A case

report. International Journal of Applied Dental Sciences. 2018; 4(1):219-224.q Abdelrahman T. Brown Jaw Tumors: challenges and outcomes. International Surgery Journal.

2017;4(11):3586.q Kumar P, Jones K. Hyperparathyroid jaw tumour syndrome. Endocrine Abstracts. 2013;4(1):219-

224.

CONCLUSIONSuch complications are rarely seen in presence of good medicalstandards and provision of advanced analytic facilities but stillcases are encountered in underdeveloped countries with poorhealth facilities. The desirable aesthetic outcomes can only beproduced with great expertise and can be achieved withbiomaterial implants to replace, reconstruct and/or augment thetissue.

Category: Bone, growth plate and mineral metabolismCORRESPONDING AUTHOR: Dr. Jaida Manzoor Email address: [email protected] Children's Hospital & Institute of Child Health, Lahore, Pakistan.

The Children’s Hospital &The Institute of Child Health

Lahore